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Radiofrequency Microtenotomy for Elbow Epicondylitis: Midterm Results
Elbow epicondylitis is a painful condition caused by overuse and development of tendon degeneration. It is one of the most common elbow problems in adults, occurring both laterally and medially. “Tennis elbow” or lateral epicondylitis is diagnosed 7 to 10 times more often than the medial form, “golfer’s elbow.”1 Although these injuries are often associated with racquet sports, activities such as bowling and weightlifting and the professions of carpentry, plumbing, and meat-cutting have been described as causes.2,3
Elbow epicondylitis is thought to be the result of multiple microtraumatic events that cause disruption of the internal structure of the tendon and degeneration of the cells and matrix.4 Lesions caused by chronic overuse are now commonly called tendinosis and are not considered inflammatory in nature. Although the term tendinitis is used frequently and indiscriminately, histopathologic studies have shown that specimens of tendon obtained from areas of chronic overuse do not contain large numbers of macrophages, lymphocytes, or neutrophils.5 Rather, tendinosis appears to be a degenerative process that is characterized by the presence of dense populations of fibroblasts, vascular hyperplasia, and disorganized collagen. This constellation of findings has been termed by some authors as angiofibroblastic hyperplasia.6
Conservative care for the treatment of chronic tendinosis has been well described and is often successful. Treatment consists of rest, ice, compression, and elevation in the acute phase. This can be followed with bracing, activity modification, physical therapy, oral nonsteroidal anti-inflammatory drugs, topical applications, and injections of cortisone or platelet-rich plasma. When conservative treatment fails, surgical intervention may be considered. Procedures for the treatment of lateral epicondylitis include open débridement and release, arthroscopic débridement, percutaneous release, and radiofrequency (RF) coblation. The goals of operative treatment are to resect pathological material, to stimulate neovascularization by producing focused local bleeding, and to create a healthy scar while doing the least possible structural damage to surrounding tissues.4
The efficacy of a bipolar RF-based approach for using microtenotomy was first recognized when researchers studied the effects of transmyocardial revascularization for treating congestive heart failure.7 The use of RF- and laser-based transmyocardial revascularization initiated an angiogenic response in degenerated (ischemic) heart tissue. This success led to investigating the use of a RF-based approach for performing microtenotomy. Preclinical studies demonstrated that RF-based microtenotomy was effective for stimulating an angiogenic-healing response in tendon tissue.8 Histologic evaluation of treated tendons showed an early inflammatory response, with new blood-vessel formation by 28 days. In 2005, short-term results of this technique were published.9 This preliminary prospective case series showed that the treatment was safe and effectively improved or eliminated clinical symptoms.9 In the present midterm study, we hypothesized that pain scores would improve after RF microtenotomy and that these favorable results would continue to be observed over a longer term postoperatively.
Materials and Methods
Patients
This was a prospective, nonrandomized, single-center clinical study. After receiving institutional review board approval, patients who were 18 to 65 years of age with a diagnosis of tendinosis were approached for enrollment. For inclusion, patients had to be symptomatic for at least 6 months and had to have failed extensive conservative treatments. Nonoperative treatment included activity modification, enrollment in a facility- or home-based exercise program, bracing, oral nonsteroidal anti-inflammatory medication, and cortisone injection. Candidates with diabetes, confirmed or suspected pregnancy, surgery in the same tendon, implanted hardware adjacent to the target treatment region, or who were receiving care under workers’ compensation or had litigation-related injury were excluded. A single clinician performed a thorough medical history and clinical evaluation. The clinical follow-up and data collection were performed by an independent medical technician.
Clinical Outcomes
Pain status was assessed by using a visual analog scale (VAS). Postoperative clinical assessment was conducted within the first 2 days; at 7 to 10 days; at 4 to 6 weeks; and at 3, 6, 12, and 24 months, up to 9 years postoperatively. The VAS scales were completed annually up to 9 years after the procedure.
The percent improvement of VAS score was calculated. This value represented the difference between the patient’s preoperative and most recent VAS assessments. Failure of the procedure was defined as less than 50% improvement of the VAS score.
The RF-Based Microtenotomy Device
The Topaz Microdebrider (ArthroCare), connected to a System 2000 generator at setting 4 (175 V-RMS), was used to perform the RF-based microtenotomy. The device uses a controlled plasma-mediated RF-based process (coblation). Radiofrequency energy is used to excite the electrolytes in a conductive medium, such as a saline solution, to create precisely focused plasma. The energized particles in the plasma have sufficient energy to break molecular bonds,10,11 excising or dissolving (ie, ablating) soft tissue at relatively low temperatures (typically, 40°-70° C).12,13 The diameter of the active tip of the Topaz device is 0.8 mm.
Surgical Procedure
The senior author performed the majority of procedures in this study. Near the end of the series, the senior author’s associate also performed procedures. The symptomatic area of the tendon was identified and marked while the patient was alert. After the patient was positioned appropriately, light sedation was administered. A tourniquet was placed over the treatment limb and inflated to 250 mm Hg. A small incision, approximately 3 cm in length, was made over the marked treatment site to expose the involved tendon. After initiating sterile isotonic saline flow of 1 drop every 1 to 2 seconds from a line connected to the RF system, the tip of the device was placed on the tendon perpendicular to its surface (Figure 1). Using a light touch, it was activated for 500 milliseconds using a timer accessory for the control box. Five to 8 grams of pressure were applied with the device to penetrate the tendon and achieve successful ablation. The RF applications were performed at 5-mm intervals, to create a grid-like pattern on and throughout the symptomatic tendon area. The tendon was perforated to a depth of several millimeters on every second or third application throughout the treatment grid. After treatment of the symptomatic area, the wound was irrigated with copious amounts of normal saline solution and closed with interrupted nylon suture. Local anesthetic was injected only in the skin and in subcutaneous tissue. Standard wound dressings were applied. In the immediate postoperative period, the patient was advised to begin gentle active and passive range-of-motion exercises. Each patient was evaluated at 1 week postoperatively. At 6 weeks, patients were permitted to increase the intensity of their activities. Return to sports and heavy lifting was allowed once the patient was asymptomatic and had achieved full strength and range of motion; this typically occurred at 6 to 9 weeks after surgery.
Statistical Analysis
Normally distributed data were described using standard parametric statistics (ie, mean and standard deviation); non-normally distributed data were characterized using nonparametric descriptors (ie, median and quartiles). Statistical evaluation of improvement in pain status was performed by calculating 99% confidence intervals and using the Student t test for change between subsequent time points. Use of confidence intervals provides a descriptive analysis of the observed treatment effect, while permitting determination of statistical relevance. In all statistical testing, confidence bounds not including 0 were considered statistically significant. Probability of P ≤ .01 for committing type I experiment-wise error (rejecting a true null hypothesis) was selected for all statistical testing because of our lack of a control group, small sample size, and evaluation of multiple postoperative time points.
Results
Eighty consecutive patients with tendinosis of the elbow were included in this study. Sixty-nine patients were treated for lateral epicondylitis and 11 for medial epicondylitis. The average age of the patients (33 women, 47 men) was 50 years. The duration of follow-up evaluation ranged from 6 months to 9 years (mean, 2.5 years; median, 2 years). The Table presents the VAS improvement for these patients after the RF microtenotomy.
Within the lateral epicondylitis group, 91% (63/69) of the patients reported a successful outcome. The postoperative VAS improved to 1.3 from 6.9, which demonstrated an 81% improvement. Of the 6 patients that did not improve, 2 underwent repeat surgery.
Among the patients treated for medial epicondylitis, 91% (10/11) reported improvement in symptoms. The postoperative VAS improved to 1.3 from 6.1, a 79% improvement. One patient did not improve and did not undergo repeat surgery.
Discussion
For the treatment of medial and lateral elbow epicondylitis, RF microtenotomy is successful in 91% of patients. Symptomatic improvement was observed up to 9 years postoperatively. During this study, no complications were recorded; 7 treatment failures occurred. When compared with other techniques, the results with RF microtenotomy are equivalent or better.
In a retrospective study, Szabo and colleagues14 compared open, arthroscopic, and percutaneous release for lateral elbow tendinosis. They found the 3 methods to be highly effective for the treatment of tendinosis with no significant difference between them. Resection of the epicondyle and transfer of the anconeus muscle was found to be effective (94%) in a retrospective study by Almquist and colleagues.15 Dunn and coauthors16 reported a 97% success rate at 10 to 14 years postoperatively with a mini-open technique. Rubenthaler and colleagues17 showed 88% effectiveness for the open technique and 93% for the arthroscopic technique. With arthroscopic release of the extensor carpi radialis brevis tendon, Lattermann and coauthors18 reported clinical improvement in 94% of patients. In a study by Rose and colleagues,19 denervation of the lateral epicondyle was effective in relieving pain in 80% of patients who had had a positive response to a local anesthetic block. In a recently published study by Koh and coauthors,20 19 of 20 patients experienced a favorable outcome after treatment with ultrasonic microresection.
Regardless of surgical methods and their reported success rate, complications are associated with elbow surgery. Postoperative problems may include restricted function, elbow instability, persistent muscle weakness, and painful neuroma of the posterior cutaneous nerve.10,21,22 The recent introduction of arthroscopic release offers the potential for less morbidity and enables visualization of the elbow joint. However, disadvantages of the arthroscopic approach include violation of the joint for extra-articular pathology, increased operative time and cost, and neurovascular complications. Additionally, it is possible that the entire spectrum of extra-articular tendinosis cannot be effectively identified arthroscopically.23 In a prospective, randomized study, Meknas and colleagues24 compared RF microtenotomy with extensor tendon release and repair. They showed that patients treated with RF-microtenotomy experienced earlier pain relief and improved grip strength over the release group.
Different proposed mechanisms of action have been described to explain the favorable effects of the RF-based microtenotomy procedure, such as induced healing by an angiogenic response in the tendon tissue. In an animal study, Harwood and colleagues8 showed that low-dose RF-based plasma microtenotomy has the ability to stimulate angiogenic growth factors in tendons, such as αv integrin and vascular endothelial growth factor. These factors have been shown to be associated with healing.8 Early inflammatory response with new-vessel formation after 28 days was found in another animal study using the same method.25 Evaluation of RF-based methods in a prospective controlled laboratory study using a rabbit-tendon model showed histologic evidence of early inflammation with development of neovasculature after treatment.8 A later histologic study using an aged Achilles rabbit tendon model was performed to evaluate the effect of RF-based plasma microtenotomy on collagen remodeling.25 The degenerated tendon showed gaps, few normal crimpings, and a lack of reflectivity under polarized light. At 9 days after treatment, the treated tendon showed localized irregular crimpings, and, at 30 days, it showed regular crimping, tightly dense collagen fibers, and hypercellularity with good reflectivity. This was similar in appearance to a normal nondegenerated tendon (Figures 2A-2D). The RF-treated tendon also demonstrated an increase in production of insulin-like growth factor-1, β-fibroblast growth factor-1, αv integrin, and vascular endothelial growth factor.
Pathologic nerve ingrowth or nerve irritation in the tendon substance has been considered a possible cause of the pain experienced with tendinosis. Radiofrequency treatment has been shown to induce acute degeneration and ablation of sensory nerve fibers.26 These degenerated nerve fibers were observed to regenerate at 90 days after treatment.27 These findings provide potential evidence for early pain relief that is maintained long term as the nerves regenerate.
This midterm follow-up of patients with elbow epicondylitis has shown that RF-based microtenotomy can produce successful, durable results. Microtenotomy is a technically simple procedure to perform and is associated with a rapid and uncomplicated recovery. It is safe and can effectively eliminate or markedly reduce clinical symptoms.
Limitations
Lateral epicondylitis has been described as a self-limited disease, with resolution of symptoms at 12 to 18 months with conservative treatment. This perspective challenges the indication of any proposed surgical treatment for the condition. Although the results of this research demonstrated the benefits of RF microtenotomy, there are inherent limitations of the study design. The study lacks a control group, and randomization would improve the strength of the study. Additional outcome measures, such as Disabilities of the Arm, Shoulder, and Hand score, and grip strength could complement pain scores to provide more data. These data were collected in a preliminary study.9 Postoperative histologic analysis of treated human tissue would be ideal, but ethical considerations limit study to animal models. An additional limitation is potential examiner bias. Data collection was performed by an independent medical technician; a third-party blinded evaluation could have been performed, but this was not feasible in a clinical setting.
Conclusion
Radiofrequency-based microtenotomy is a safe and effective procedure for elbow epicondylitis. The results are durable with successful outcomes observed 9 years after surgery.
1. Leach RE, Miller JK. Lateral and medial epicondylitis of the elbow. Clin Sports Med. 1987;6(2):259-272.
2. Vangsness CT Jr, Jobe FW. Surgical technique of medial epicondylitis: Results in 35 elbows. J Bone Joint Surg Br. 1991;73(3):409-411.
3. Galloway M, DeMaio M, Mangine R. Rehabilitative techniques in the treatment of medial and lateral epicondylitis. Orthopedics. 1992;15(9):1089-1096.
4. Kraushaar BS, Nirschl RP. Tendinosis of the elbow (tennis elbow). Clinical features and findings of histological, immunohistochemical, and electron microscopy studies. J Bone Joint Surg Am. 1999;81(2):259-278.
5. Leadbetter WB. Cell-matrix response in tendon injury. Clin Sports Med. 1992;11(3):533-578.
6. Nirschl RP. Tennis elbow tendinosis: pathoanatomy, nonsurgical and surgical management. In: Fine LJ, ed. Repetitive Motion Disorders of the Upper Extremity. Rosemont, IL: American Academy of Orthopaedic Surgeons; 1995:467-479.
7. Chu V, Kuang J, Aiaid A, Korkola S, Chiu RC. Angiogenic response induced by mechanical transmyocardial revascularization. J Thorac Cardiovasc Surg 1999;118:849-856.
8. Harwood R, Bowden K, Amiel M, Tasto JP, Amiel D. Structural and angiogenic response to bipolar radiofrequency treatment of normal rabbit achilles tendon: a potential application to the treatment of tendinosis. Trans Orthop Res Soc. 2003;28:819.
9. Tasto JP, Cummings J, Medlock V, Hardesty R, Amiel D. Microtenotomy using a radiofrequency probe to treat lateral epicondylitis. Arthroscopy. 2005;21(7):851-860.
10. Woloszko J, Stalder KR, Brown IG. Plasma characteristics of repetitively-pulsed electrical discharges in saline solutions used for surgical procedures. IEEE Trans Plasma Sci. 2002;30:1376-1383.
11. Stalder KR, Woloszko J, Brown IG, Smith CD. Repetitive plasma discharges in saline solutions. Appl Phys Lett. 2001;79:4503-4505.
12. Woloszko J, Gilbride C. Coblation technology (plasma mediated ablation for otolaryngology applications). Proc SPIE. 2000;3907:306–316.
13. Woloszko J, Kwende MM, Stalder KR. Coblation in otolaryngology. Proc SPIE. 2003;4949:341–352.
14. Szabo SJ, Savoie FH 3rd, Field LD, Ramsey JR, Hosemann CD. Tendinosis of the extensor carpi radialis brevis: an evaluation of three methods of operative treatment. J Shoulder Elbow Surg Am. 2006;15(6):721-727.
15. Almquist EE, Necking L, Bach AW. Epicondylar resection with anconeus transfer for chronic lateral epicondylitis. J Hand Surg Am. 1998;23(4):723-731.
16. Dunn JH, Kim JJ, Davis L, Nirschl RP. Ten- to 14-year follow-up of the Nirschl surgical technique for lateral epicondylitis. Am J Sports Med. 2008;36(2):261-266.
17. Rubenthaler F, Wiese M, Senge A, Keller L, Wittenberg RH. Long-term follow-up of open and endoscopic Hohmann procedures for lateral epicondylitis. Arthroscopy. 2005;21(6):684-690.
18. Lattermann C, Romeo AA, Anbari A, et al. Arthroscopic debridement of the extensor carpi radialis brevis for the treatment of recalcitrant lateral epicondylitis. J Shoulder Elbow Surg. 2010;19(5):651-656.
19. Rose NE, Forman SK, Dellon AL. Denervation of the lateral epicondyle for treatment of chronic lateral epicondylitis. J Hand Surg Am. 2013;38(2):344-349.
20. Koh JS, Mohan PC, Howe TS, et al. Fasciotomy and surgical tenotomy for recalcitrant lateral elbow tendonopathy: early clinical experience with a novel device for minimally invasive percutaneous microresection. Am J Sports Med. 2013;41(3):636-644.
21. Nirschl RP, Ashman ES. Elbow tendonopathy: tennis elbow. Clin Sports Med. 2003;22(4):813-836.
22. Dellon AL, Kim J, Ducic I. Painful neuroma of the posterior cutaneous nerve of the forearm after surgery for lateral humeral epicondylitis. J Hand Surg Am. 2004;29(3):387-390.
23. Cummins CA. Lateral epicondylitis: in-vivo assessment of arthroscopic debridement and correlation with patient outcomes. Am J Sports Med. 2006;34(9):1486-1491.
24. Meknas K, Odden-Miland A, Mercer JB, Castillejo M, Johansen O. Radiofrequency microtenotomy: a promising method for treatment of recalcitrant lateral epicondylitis. Am J Sports Med. 2008;36(10):1960-1965.
25. Takahashi N, Tasto JP, Locke J, et al. The use of radiofrequency (RF) for the treatment of chronic tendinosis. Paper presented at: 6th Biennial Congress of the International Society of Arthroscopy, Knee Surgery, and Orthopaedic Sports Medicine Congress; May 2007; Florence, Italy. Abstract 1433.
26. Takahashi N, Tasto JP, Ritter M, et al. Pain relief through an antinociceptive effect after radiofrequency application. Am J Sports Med. 2007;35(5):805-810.
27. Ochiai N, Tasto JP, Ohtori S, Takahashi N, Moriya H, Amiel D. Nerve regeneration after radiofrequency ablation. Am J Sports Med. 2007;35(11):1940-1944.
Elbow epicondylitis is a painful condition caused by overuse and development of tendon degeneration. It is one of the most common elbow problems in adults, occurring both laterally and medially. “Tennis elbow” or lateral epicondylitis is diagnosed 7 to 10 times more often than the medial form, “golfer’s elbow.”1 Although these injuries are often associated with racquet sports, activities such as bowling and weightlifting and the professions of carpentry, plumbing, and meat-cutting have been described as causes.2,3
Elbow epicondylitis is thought to be the result of multiple microtraumatic events that cause disruption of the internal structure of the tendon and degeneration of the cells and matrix.4 Lesions caused by chronic overuse are now commonly called tendinosis and are not considered inflammatory in nature. Although the term tendinitis is used frequently and indiscriminately, histopathologic studies have shown that specimens of tendon obtained from areas of chronic overuse do not contain large numbers of macrophages, lymphocytes, or neutrophils.5 Rather, tendinosis appears to be a degenerative process that is characterized by the presence of dense populations of fibroblasts, vascular hyperplasia, and disorganized collagen. This constellation of findings has been termed by some authors as angiofibroblastic hyperplasia.6
Conservative care for the treatment of chronic tendinosis has been well described and is often successful. Treatment consists of rest, ice, compression, and elevation in the acute phase. This can be followed with bracing, activity modification, physical therapy, oral nonsteroidal anti-inflammatory drugs, topical applications, and injections of cortisone or platelet-rich plasma. When conservative treatment fails, surgical intervention may be considered. Procedures for the treatment of lateral epicondylitis include open débridement and release, arthroscopic débridement, percutaneous release, and radiofrequency (RF) coblation. The goals of operative treatment are to resect pathological material, to stimulate neovascularization by producing focused local bleeding, and to create a healthy scar while doing the least possible structural damage to surrounding tissues.4
The efficacy of a bipolar RF-based approach for using microtenotomy was first recognized when researchers studied the effects of transmyocardial revascularization for treating congestive heart failure.7 The use of RF- and laser-based transmyocardial revascularization initiated an angiogenic response in degenerated (ischemic) heart tissue. This success led to investigating the use of a RF-based approach for performing microtenotomy. Preclinical studies demonstrated that RF-based microtenotomy was effective for stimulating an angiogenic-healing response in tendon tissue.8 Histologic evaluation of treated tendons showed an early inflammatory response, with new blood-vessel formation by 28 days. In 2005, short-term results of this technique were published.9 This preliminary prospective case series showed that the treatment was safe and effectively improved or eliminated clinical symptoms.9 In the present midterm study, we hypothesized that pain scores would improve after RF microtenotomy and that these favorable results would continue to be observed over a longer term postoperatively.
Materials and Methods
Patients
This was a prospective, nonrandomized, single-center clinical study. After receiving institutional review board approval, patients who were 18 to 65 years of age with a diagnosis of tendinosis were approached for enrollment. For inclusion, patients had to be symptomatic for at least 6 months and had to have failed extensive conservative treatments. Nonoperative treatment included activity modification, enrollment in a facility- or home-based exercise program, bracing, oral nonsteroidal anti-inflammatory medication, and cortisone injection. Candidates with diabetes, confirmed or suspected pregnancy, surgery in the same tendon, implanted hardware adjacent to the target treatment region, or who were receiving care under workers’ compensation or had litigation-related injury were excluded. A single clinician performed a thorough medical history and clinical evaluation. The clinical follow-up and data collection were performed by an independent medical technician.
Clinical Outcomes
Pain status was assessed by using a visual analog scale (VAS). Postoperative clinical assessment was conducted within the first 2 days; at 7 to 10 days; at 4 to 6 weeks; and at 3, 6, 12, and 24 months, up to 9 years postoperatively. The VAS scales were completed annually up to 9 years after the procedure.
The percent improvement of VAS score was calculated. This value represented the difference between the patient’s preoperative and most recent VAS assessments. Failure of the procedure was defined as less than 50% improvement of the VAS score.
The RF-Based Microtenotomy Device
The Topaz Microdebrider (ArthroCare), connected to a System 2000 generator at setting 4 (175 V-RMS), was used to perform the RF-based microtenotomy. The device uses a controlled plasma-mediated RF-based process (coblation). Radiofrequency energy is used to excite the electrolytes in a conductive medium, such as a saline solution, to create precisely focused plasma. The energized particles in the plasma have sufficient energy to break molecular bonds,10,11 excising or dissolving (ie, ablating) soft tissue at relatively low temperatures (typically, 40°-70° C).12,13 The diameter of the active tip of the Topaz device is 0.8 mm.
Surgical Procedure
The senior author performed the majority of procedures in this study. Near the end of the series, the senior author’s associate also performed procedures. The symptomatic area of the tendon was identified and marked while the patient was alert. After the patient was positioned appropriately, light sedation was administered. A tourniquet was placed over the treatment limb and inflated to 250 mm Hg. A small incision, approximately 3 cm in length, was made over the marked treatment site to expose the involved tendon. After initiating sterile isotonic saline flow of 1 drop every 1 to 2 seconds from a line connected to the RF system, the tip of the device was placed on the tendon perpendicular to its surface (Figure 1). Using a light touch, it was activated for 500 milliseconds using a timer accessory for the control box. Five to 8 grams of pressure were applied with the device to penetrate the tendon and achieve successful ablation. The RF applications were performed at 5-mm intervals, to create a grid-like pattern on and throughout the symptomatic tendon area. The tendon was perforated to a depth of several millimeters on every second or third application throughout the treatment grid. After treatment of the symptomatic area, the wound was irrigated with copious amounts of normal saline solution and closed with interrupted nylon suture. Local anesthetic was injected only in the skin and in subcutaneous tissue. Standard wound dressings were applied. In the immediate postoperative period, the patient was advised to begin gentle active and passive range-of-motion exercises. Each patient was evaluated at 1 week postoperatively. At 6 weeks, patients were permitted to increase the intensity of their activities. Return to sports and heavy lifting was allowed once the patient was asymptomatic and had achieved full strength and range of motion; this typically occurred at 6 to 9 weeks after surgery.
Statistical Analysis
Normally distributed data were described using standard parametric statistics (ie, mean and standard deviation); non-normally distributed data were characterized using nonparametric descriptors (ie, median and quartiles). Statistical evaluation of improvement in pain status was performed by calculating 99% confidence intervals and using the Student t test for change between subsequent time points. Use of confidence intervals provides a descriptive analysis of the observed treatment effect, while permitting determination of statistical relevance. In all statistical testing, confidence bounds not including 0 were considered statistically significant. Probability of P ≤ .01 for committing type I experiment-wise error (rejecting a true null hypothesis) was selected for all statistical testing because of our lack of a control group, small sample size, and evaluation of multiple postoperative time points.
Results
Eighty consecutive patients with tendinosis of the elbow were included in this study. Sixty-nine patients were treated for lateral epicondylitis and 11 for medial epicondylitis. The average age of the patients (33 women, 47 men) was 50 years. The duration of follow-up evaluation ranged from 6 months to 9 years (mean, 2.5 years; median, 2 years). The Table presents the VAS improvement for these patients after the RF microtenotomy.
Within the lateral epicondylitis group, 91% (63/69) of the patients reported a successful outcome. The postoperative VAS improved to 1.3 from 6.9, which demonstrated an 81% improvement. Of the 6 patients that did not improve, 2 underwent repeat surgery.
Among the patients treated for medial epicondylitis, 91% (10/11) reported improvement in symptoms. The postoperative VAS improved to 1.3 from 6.1, a 79% improvement. One patient did not improve and did not undergo repeat surgery.
Discussion
For the treatment of medial and lateral elbow epicondylitis, RF microtenotomy is successful in 91% of patients. Symptomatic improvement was observed up to 9 years postoperatively. During this study, no complications were recorded; 7 treatment failures occurred. When compared with other techniques, the results with RF microtenotomy are equivalent or better.
In a retrospective study, Szabo and colleagues14 compared open, arthroscopic, and percutaneous release for lateral elbow tendinosis. They found the 3 methods to be highly effective for the treatment of tendinosis with no significant difference between them. Resection of the epicondyle and transfer of the anconeus muscle was found to be effective (94%) in a retrospective study by Almquist and colleagues.15 Dunn and coauthors16 reported a 97% success rate at 10 to 14 years postoperatively with a mini-open technique. Rubenthaler and colleagues17 showed 88% effectiveness for the open technique and 93% for the arthroscopic technique. With arthroscopic release of the extensor carpi radialis brevis tendon, Lattermann and coauthors18 reported clinical improvement in 94% of patients. In a study by Rose and colleagues,19 denervation of the lateral epicondyle was effective in relieving pain in 80% of patients who had had a positive response to a local anesthetic block. In a recently published study by Koh and coauthors,20 19 of 20 patients experienced a favorable outcome after treatment with ultrasonic microresection.
Regardless of surgical methods and their reported success rate, complications are associated with elbow surgery. Postoperative problems may include restricted function, elbow instability, persistent muscle weakness, and painful neuroma of the posterior cutaneous nerve.10,21,22 The recent introduction of arthroscopic release offers the potential for less morbidity and enables visualization of the elbow joint. However, disadvantages of the arthroscopic approach include violation of the joint for extra-articular pathology, increased operative time and cost, and neurovascular complications. Additionally, it is possible that the entire spectrum of extra-articular tendinosis cannot be effectively identified arthroscopically.23 In a prospective, randomized study, Meknas and colleagues24 compared RF microtenotomy with extensor tendon release and repair. They showed that patients treated with RF-microtenotomy experienced earlier pain relief and improved grip strength over the release group.
Different proposed mechanisms of action have been described to explain the favorable effects of the RF-based microtenotomy procedure, such as induced healing by an angiogenic response in the tendon tissue. In an animal study, Harwood and colleagues8 showed that low-dose RF-based plasma microtenotomy has the ability to stimulate angiogenic growth factors in tendons, such as αv integrin and vascular endothelial growth factor. These factors have been shown to be associated with healing.8 Early inflammatory response with new-vessel formation after 28 days was found in another animal study using the same method.25 Evaluation of RF-based methods in a prospective controlled laboratory study using a rabbit-tendon model showed histologic evidence of early inflammation with development of neovasculature after treatment.8 A later histologic study using an aged Achilles rabbit tendon model was performed to evaluate the effect of RF-based plasma microtenotomy on collagen remodeling.25 The degenerated tendon showed gaps, few normal crimpings, and a lack of reflectivity under polarized light. At 9 days after treatment, the treated tendon showed localized irregular crimpings, and, at 30 days, it showed regular crimping, tightly dense collagen fibers, and hypercellularity with good reflectivity. This was similar in appearance to a normal nondegenerated tendon (Figures 2A-2D). The RF-treated tendon also demonstrated an increase in production of insulin-like growth factor-1, β-fibroblast growth factor-1, αv integrin, and vascular endothelial growth factor.
Pathologic nerve ingrowth or nerve irritation in the tendon substance has been considered a possible cause of the pain experienced with tendinosis. Radiofrequency treatment has been shown to induce acute degeneration and ablation of sensory nerve fibers.26 These degenerated nerve fibers were observed to regenerate at 90 days after treatment.27 These findings provide potential evidence for early pain relief that is maintained long term as the nerves regenerate.
This midterm follow-up of patients with elbow epicondylitis has shown that RF-based microtenotomy can produce successful, durable results. Microtenotomy is a technically simple procedure to perform and is associated with a rapid and uncomplicated recovery. It is safe and can effectively eliminate or markedly reduce clinical symptoms.
Limitations
Lateral epicondylitis has been described as a self-limited disease, with resolution of symptoms at 12 to 18 months with conservative treatment. This perspective challenges the indication of any proposed surgical treatment for the condition. Although the results of this research demonstrated the benefits of RF microtenotomy, there are inherent limitations of the study design. The study lacks a control group, and randomization would improve the strength of the study. Additional outcome measures, such as Disabilities of the Arm, Shoulder, and Hand score, and grip strength could complement pain scores to provide more data. These data were collected in a preliminary study.9 Postoperative histologic analysis of treated human tissue would be ideal, but ethical considerations limit study to animal models. An additional limitation is potential examiner bias. Data collection was performed by an independent medical technician; a third-party blinded evaluation could have been performed, but this was not feasible in a clinical setting.
Conclusion
Radiofrequency-based microtenotomy is a safe and effective procedure for elbow epicondylitis. The results are durable with successful outcomes observed 9 years after surgery.
Elbow epicondylitis is a painful condition caused by overuse and development of tendon degeneration. It is one of the most common elbow problems in adults, occurring both laterally and medially. “Tennis elbow” or lateral epicondylitis is diagnosed 7 to 10 times more often than the medial form, “golfer’s elbow.”1 Although these injuries are often associated with racquet sports, activities such as bowling and weightlifting and the professions of carpentry, plumbing, and meat-cutting have been described as causes.2,3
Elbow epicondylitis is thought to be the result of multiple microtraumatic events that cause disruption of the internal structure of the tendon and degeneration of the cells and matrix.4 Lesions caused by chronic overuse are now commonly called tendinosis and are not considered inflammatory in nature. Although the term tendinitis is used frequently and indiscriminately, histopathologic studies have shown that specimens of tendon obtained from areas of chronic overuse do not contain large numbers of macrophages, lymphocytes, or neutrophils.5 Rather, tendinosis appears to be a degenerative process that is characterized by the presence of dense populations of fibroblasts, vascular hyperplasia, and disorganized collagen. This constellation of findings has been termed by some authors as angiofibroblastic hyperplasia.6
Conservative care for the treatment of chronic tendinosis has been well described and is often successful. Treatment consists of rest, ice, compression, and elevation in the acute phase. This can be followed with bracing, activity modification, physical therapy, oral nonsteroidal anti-inflammatory drugs, topical applications, and injections of cortisone or platelet-rich plasma. When conservative treatment fails, surgical intervention may be considered. Procedures for the treatment of lateral epicondylitis include open débridement and release, arthroscopic débridement, percutaneous release, and radiofrequency (RF) coblation. The goals of operative treatment are to resect pathological material, to stimulate neovascularization by producing focused local bleeding, and to create a healthy scar while doing the least possible structural damage to surrounding tissues.4
The efficacy of a bipolar RF-based approach for using microtenotomy was first recognized when researchers studied the effects of transmyocardial revascularization for treating congestive heart failure.7 The use of RF- and laser-based transmyocardial revascularization initiated an angiogenic response in degenerated (ischemic) heart tissue. This success led to investigating the use of a RF-based approach for performing microtenotomy. Preclinical studies demonstrated that RF-based microtenotomy was effective for stimulating an angiogenic-healing response in tendon tissue.8 Histologic evaluation of treated tendons showed an early inflammatory response, with new blood-vessel formation by 28 days. In 2005, short-term results of this technique were published.9 This preliminary prospective case series showed that the treatment was safe and effectively improved or eliminated clinical symptoms.9 In the present midterm study, we hypothesized that pain scores would improve after RF microtenotomy and that these favorable results would continue to be observed over a longer term postoperatively.
Materials and Methods
Patients
This was a prospective, nonrandomized, single-center clinical study. After receiving institutional review board approval, patients who were 18 to 65 years of age with a diagnosis of tendinosis were approached for enrollment. For inclusion, patients had to be symptomatic for at least 6 months and had to have failed extensive conservative treatments. Nonoperative treatment included activity modification, enrollment in a facility- or home-based exercise program, bracing, oral nonsteroidal anti-inflammatory medication, and cortisone injection. Candidates with diabetes, confirmed or suspected pregnancy, surgery in the same tendon, implanted hardware adjacent to the target treatment region, or who were receiving care under workers’ compensation or had litigation-related injury were excluded. A single clinician performed a thorough medical history and clinical evaluation. The clinical follow-up and data collection were performed by an independent medical technician.
Clinical Outcomes
Pain status was assessed by using a visual analog scale (VAS). Postoperative clinical assessment was conducted within the first 2 days; at 7 to 10 days; at 4 to 6 weeks; and at 3, 6, 12, and 24 months, up to 9 years postoperatively. The VAS scales were completed annually up to 9 years after the procedure.
The percent improvement of VAS score was calculated. This value represented the difference between the patient’s preoperative and most recent VAS assessments. Failure of the procedure was defined as less than 50% improvement of the VAS score.
The RF-Based Microtenotomy Device
The Topaz Microdebrider (ArthroCare), connected to a System 2000 generator at setting 4 (175 V-RMS), was used to perform the RF-based microtenotomy. The device uses a controlled plasma-mediated RF-based process (coblation). Radiofrequency energy is used to excite the electrolytes in a conductive medium, such as a saline solution, to create precisely focused plasma. The energized particles in the plasma have sufficient energy to break molecular bonds,10,11 excising or dissolving (ie, ablating) soft tissue at relatively low temperatures (typically, 40°-70° C).12,13 The diameter of the active tip of the Topaz device is 0.8 mm.
Surgical Procedure
The senior author performed the majority of procedures in this study. Near the end of the series, the senior author’s associate also performed procedures. The symptomatic area of the tendon was identified and marked while the patient was alert. After the patient was positioned appropriately, light sedation was administered. A tourniquet was placed over the treatment limb and inflated to 250 mm Hg. A small incision, approximately 3 cm in length, was made over the marked treatment site to expose the involved tendon. After initiating sterile isotonic saline flow of 1 drop every 1 to 2 seconds from a line connected to the RF system, the tip of the device was placed on the tendon perpendicular to its surface (Figure 1). Using a light touch, it was activated for 500 milliseconds using a timer accessory for the control box. Five to 8 grams of pressure were applied with the device to penetrate the tendon and achieve successful ablation. The RF applications were performed at 5-mm intervals, to create a grid-like pattern on and throughout the symptomatic tendon area. The tendon was perforated to a depth of several millimeters on every second or third application throughout the treatment grid. After treatment of the symptomatic area, the wound was irrigated with copious amounts of normal saline solution and closed with interrupted nylon suture. Local anesthetic was injected only in the skin and in subcutaneous tissue. Standard wound dressings were applied. In the immediate postoperative period, the patient was advised to begin gentle active and passive range-of-motion exercises. Each patient was evaluated at 1 week postoperatively. At 6 weeks, patients were permitted to increase the intensity of their activities. Return to sports and heavy lifting was allowed once the patient was asymptomatic and had achieved full strength and range of motion; this typically occurred at 6 to 9 weeks after surgery.
Statistical Analysis
Normally distributed data were described using standard parametric statistics (ie, mean and standard deviation); non-normally distributed data were characterized using nonparametric descriptors (ie, median and quartiles). Statistical evaluation of improvement in pain status was performed by calculating 99% confidence intervals and using the Student t test for change between subsequent time points. Use of confidence intervals provides a descriptive analysis of the observed treatment effect, while permitting determination of statistical relevance. In all statistical testing, confidence bounds not including 0 were considered statistically significant. Probability of P ≤ .01 for committing type I experiment-wise error (rejecting a true null hypothesis) was selected for all statistical testing because of our lack of a control group, small sample size, and evaluation of multiple postoperative time points.
Results
Eighty consecutive patients with tendinosis of the elbow were included in this study. Sixty-nine patients were treated for lateral epicondylitis and 11 for medial epicondylitis. The average age of the patients (33 women, 47 men) was 50 years. The duration of follow-up evaluation ranged from 6 months to 9 years (mean, 2.5 years; median, 2 years). The Table presents the VAS improvement for these patients after the RF microtenotomy.
Within the lateral epicondylitis group, 91% (63/69) of the patients reported a successful outcome. The postoperative VAS improved to 1.3 from 6.9, which demonstrated an 81% improvement. Of the 6 patients that did not improve, 2 underwent repeat surgery.
Among the patients treated for medial epicondylitis, 91% (10/11) reported improvement in symptoms. The postoperative VAS improved to 1.3 from 6.1, a 79% improvement. One patient did not improve and did not undergo repeat surgery.
Discussion
For the treatment of medial and lateral elbow epicondylitis, RF microtenotomy is successful in 91% of patients. Symptomatic improvement was observed up to 9 years postoperatively. During this study, no complications were recorded; 7 treatment failures occurred. When compared with other techniques, the results with RF microtenotomy are equivalent or better.
In a retrospective study, Szabo and colleagues14 compared open, arthroscopic, and percutaneous release for lateral elbow tendinosis. They found the 3 methods to be highly effective for the treatment of tendinosis with no significant difference between them. Resection of the epicondyle and transfer of the anconeus muscle was found to be effective (94%) in a retrospective study by Almquist and colleagues.15 Dunn and coauthors16 reported a 97% success rate at 10 to 14 years postoperatively with a mini-open technique. Rubenthaler and colleagues17 showed 88% effectiveness for the open technique and 93% for the arthroscopic technique. With arthroscopic release of the extensor carpi radialis brevis tendon, Lattermann and coauthors18 reported clinical improvement in 94% of patients. In a study by Rose and colleagues,19 denervation of the lateral epicondyle was effective in relieving pain in 80% of patients who had had a positive response to a local anesthetic block. In a recently published study by Koh and coauthors,20 19 of 20 patients experienced a favorable outcome after treatment with ultrasonic microresection.
Regardless of surgical methods and their reported success rate, complications are associated with elbow surgery. Postoperative problems may include restricted function, elbow instability, persistent muscle weakness, and painful neuroma of the posterior cutaneous nerve.10,21,22 The recent introduction of arthroscopic release offers the potential for less morbidity and enables visualization of the elbow joint. However, disadvantages of the arthroscopic approach include violation of the joint for extra-articular pathology, increased operative time and cost, and neurovascular complications. Additionally, it is possible that the entire spectrum of extra-articular tendinosis cannot be effectively identified arthroscopically.23 In a prospective, randomized study, Meknas and colleagues24 compared RF microtenotomy with extensor tendon release and repair. They showed that patients treated with RF-microtenotomy experienced earlier pain relief and improved grip strength over the release group.
Different proposed mechanisms of action have been described to explain the favorable effects of the RF-based microtenotomy procedure, such as induced healing by an angiogenic response in the tendon tissue. In an animal study, Harwood and colleagues8 showed that low-dose RF-based plasma microtenotomy has the ability to stimulate angiogenic growth factors in tendons, such as αv integrin and vascular endothelial growth factor. These factors have been shown to be associated with healing.8 Early inflammatory response with new-vessel formation after 28 days was found in another animal study using the same method.25 Evaluation of RF-based methods in a prospective controlled laboratory study using a rabbit-tendon model showed histologic evidence of early inflammation with development of neovasculature after treatment.8 A later histologic study using an aged Achilles rabbit tendon model was performed to evaluate the effect of RF-based plasma microtenotomy on collagen remodeling.25 The degenerated tendon showed gaps, few normal crimpings, and a lack of reflectivity under polarized light. At 9 days after treatment, the treated tendon showed localized irregular crimpings, and, at 30 days, it showed regular crimping, tightly dense collagen fibers, and hypercellularity with good reflectivity. This was similar in appearance to a normal nondegenerated tendon (Figures 2A-2D). The RF-treated tendon also demonstrated an increase in production of insulin-like growth factor-1, β-fibroblast growth factor-1, αv integrin, and vascular endothelial growth factor.
Pathologic nerve ingrowth or nerve irritation in the tendon substance has been considered a possible cause of the pain experienced with tendinosis. Radiofrequency treatment has been shown to induce acute degeneration and ablation of sensory nerve fibers.26 These degenerated nerve fibers were observed to regenerate at 90 days after treatment.27 These findings provide potential evidence for early pain relief that is maintained long term as the nerves regenerate.
This midterm follow-up of patients with elbow epicondylitis has shown that RF-based microtenotomy can produce successful, durable results. Microtenotomy is a technically simple procedure to perform and is associated with a rapid and uncomplicated recovery. It is safe and can effectively eliminate or markedly reduce clinical symptoms.
Limitations
Lateral epicondylitis has been described as a self-limited disease, with resolution of symptoms at 12 to 18 months with conservative treatment. This perspective challenges the indication of any proposed surgical treatment for the condition. Although the results of this research demonstrated the benefits of RF microtenotomy, there are inherent limitations of the study design. The study lacks a control group, and randomization would improve the strength of the study. Additional outcome measures, such as Disabilities of the Arm, Shoulder, and Hand score, and grip strength could complement pain scores to provide more data. These data were collected in a preliminary study.9 Postoperative histologic analysis of treated human tissue would be ideal, but ethical considerations limit study to animal models. An additional limitation is potential examiner bias. Data collection was performed by an independent medical technician; a third-party blinded evaluation could have been performed, but this was not feasible in a clinical setting.
Conclusion
Radiofrequency-based microtenotomy is a safe and effective procedure for elbow epicondylitis. The results are durable with successful outcomes observed 9 years after surgery.
1. Leach RE, Miller JK. Lateral and medial epicondylitis of the elbow. Clin Sports Med. 1987;6(2):259-272.
2. Vangsness CT Jr, Jobe FW. Surgical technique of medial epicondylitis: Results in 35 elbows. J Bone Joint Surg Br. 1991;73(3):409-411.
3. Galloway M, DeMaio M, Mangine R. Rehabilitative techniques in the treatment of medial and lateral epicondylitis. Orthopedics. 1992;15(9):1089-1096.
4. Kraushaar BS, Nirschl RP. Tendinosis of the elbow (tennis elbow). Clinical features and findings of histological, immunohistochemical, and electron microscopy studies. J Bone Joint Surg Am. 1999;81(2):259-278.
5. Leadbetter WB. Cell-matrix response in tendon injury. Clin Sports Med. 1992;11(3):533-578.
6. Nirschl RP. Tennis elbow tendinosis: pathoanatomy, nonsurgical and surgical management. In: Fine LJ, ed. Repetitive Motion Disorders of the Upper Extremity. Rosemont, IL: American Academy of Orthopaedic Surgeons; 1995:467-479.
7. Chu V, Kuang J, Aiaid A, Korkola S, Chiu RC. Angiogenic response induced by mechanical transmyocardial revascularization. J Thorac Cardiovasc Surg 1999;118:849-856.
8. Harwood R, Bowden K, Amiel M, Tasto JP, Amiel D. Structural and angiogenic response to bipolar radiofrequency treatment of normal rabbit achilles tendon: a potential application to the treatment of tendinosis. Trans Orthop Res Soc. 2003;28:819.
9. Tasto JP, Cummings J, Medlock V, Hardesty R, Amiel D. Microtenotomy using a radiofrequency probe to treat lateral epicondylitis. Arthroscopy. 2005;21(7):851-860.
10. Woloszko J, Stalder KR, Brown IG. Plasma characteristics of repetitively-pulsed electrical discharges in saline solutions used for surgical procedures. IEEE Trans Plasma Sci. 2002;30:1376-1383.
11. Stalder KR, Woloszko J, Brown IG, Smith CD. Repetitive plasma discharges in saline solutions. Appl Phys Lett. 2001;79:4503-4505.
12. Woloszko J, Gilbride C. Coblation technology (plasma mediated ablation for otolaryngology applications). Proc SPIE. 2000;3907:306–316.
13. Woloszko J, Kwende MM, Stalder KR. Coblation in otolaryngology. Proc SPIE. 2003;4949:341–352.
14. Szabo SJ, Savoie FH 3rd, Field LD, Ramsey JR, Hosemann CD. Tendinosis of the extensor carpi radialis brevis: an evaluation of three methods of operative treatment. J Shoulder Elbow Surg Am. 2006;15(6):721-727.
15. Almquist EE, Necking L, Bach AW. Epicondylar resection with anconeus transfer for chronic lateral epicondylitis. J Hand Surg Am. 1998;23(4):723-731.
16. Dunn JH, Kim JJ, Davis L, Nirschl RP. Ten- to 14-year follow-up of the Nirschl surgical technique for lateral epicondylitis. Am J Sports Med. 2008;36(2):261-266.
17. Rubenthaler F, Wiese M, Senge A, Keller L, Wittenberg RH. Long-term follow-up of open and endoscopic Hohmann procedures for lateral epicondylitis. Arthroscopy. 2005;21(6):684-690.
18. Lattermann C, Romeo AA, Anbari A, et al. Arthroscopic debridement of the extensor carpi radialis brevis for the treatment of recalcitrant lateral epicondylitis. J Shoulder Elbow Surg. 2010;19(5):651-656.
19. Rose NE, Forman SK, Dellon AL. Denervation of the lateral epicondyle for treatment of chronic lateral epicondylitis. J Hand Surg Am. 2013;38(2):344-349.
20. Koh JS, Mohan PC, Howe TS, et al. Fasciotomy and surgical tenotomy for recalcitrant lateral elbow tendonopathy: early clinical experience with a novel device for minimally invasive percutaneous microresection. Am J Sports Med. 2013;41(3):636-644.
21. Nirschl RP, Ashman ES. Elbow tendonopathy: tennis elbow. Clin Sports Med. 2003;22(4):813-836.
22. Dellon AL, Kim J, Ducic I. Painful neuroma of the posterior cutaneous nerve of the forearm after surgery for lateral humeral epicondylitis. J Hand Surg Am. 2004;29(3):387-390.
23. Cummins CA. Lateral epicondylitis: in-vivo assessment of arthroscopic debridement and correlation with patient outcomes. Am J Sports Med. 2006;34(9):1486-1491.
24. Meknas K, Odden-Miland A, Mercer JB, Castillejo M, Johansen O. Radiofrequency microtenotomy: a promising method for treatment of recalcitrant lateral epicondylitis. Am J Sports Med. 2008;36(10):1960-1965.
25. Takahashi N, Tasto JP, Locke J, et al. The use of radiofrequency (RF) for the treatment of chronic tendinosis. Paper presented at: 6th Biennial Congress of the International Society of Arthroscopy, Knee Surgery, and Orthopaedic Sports Medicine Congress; May 2007; Florence, Italy. Abstract 1433.
26. Takahashi N, Tasto JP, Ritter M, et al. Pain relief through an antinociceptive effect after radiofrequency application. Am J Sports Med. 2007;35(5):805-810.
27. Ochiai N, Tasto JP, Ohtori S, Takahashi N, Moriya H, Amiel D. Nerve regeneration after radiofrequency ablation. Am J Sports Med. 2007;35(11):1940-1944.
1. Leach RE, Miller JK. Lateral and medial epicondylitis of the elbow. Clin Sports Med. 1987;6(2):259-272.
2. Vangsness CT Jr, Jobe FW. Surgical technique of medial epicondylitis: Results in 35 elbows. J Bone Joint Surg Br. 1991;73(3):409-411.
3. Galloway M, DeMaio M, Mangine R. Rehabilitative techniques in the treatment of medial and lateral epicondylitis. Orthopedics. 1992;15(9):1089-1096.
4. Kraushaar BS, Nirschl RP. Tendinosis of the elbow (tennis elbow). Clinical features and findings of histological, immunohistochemical, and electron microscopy studies. J Bone Joint Surg Am. 1999;81(2):259-278.
5. Leadbetter WB. Cell-matrix response in tendon injury. Clin Sports Med. 1992;11(3):533-578.
6. Nirschl RP. Tennis elbow tendinosis: pathoanatomy, nonsurgical and surgical management. In: Fine LJ, ed. Repetitive Motion Disorders of the Upper Extremity. Rosemont, IL: American Academy of Orthopaedic Surgeons; 1995:467-479.
7. Chu V, Kuang J, Aiaid A, Korkola S, Chiu RC. Angiogenic response induced by mechanical transmyocardial revascularization. J Thorac Cardiovasc Surg 1999;118:849-856.
8. Harwood R, Bowden K, Amiel M, Tasto JP, Amiel D. Structural and angiogenic response to bipolar radiofrequency treatment of normal rabbit achilles tendon: a potential application to the treatment of tendinosis. Trans Orthop Res Soc. 2003;28:819.
9. Tasto JP, Cummings J, Medlock V, Hardesty R, Amiel D. Microtenotomy using a radiofrequency probe to treat lateral epicondylitis. Arthroscopy. 2005;21(7):851-860.
10. Woloszko J, Stalder KR, Brown IG. Plasma characteristics of repetitively-pulsed electrical discharges in saline solutions used for surgical procedures. IEEE Trans Plasma Sci. 2002;30:1376-1383.
11. Stalder KR, Woloszko J, Brown IG, Smith CD. Repetitive plasma discharges in saline solutions. Appl Phys Lett. 2001;79:4503-4505.
12. Woloszko J, Gilbride C. Coblation technology (plasma mediated ablation for otolaryngology applications). Proc SPIE. 2000;3907:306–316.
13. Woloszko J, Kwende MM, Stalder KR. Coblation in otolaryngology. Proc SPIE. 2003;4949:341–352.
14. Szabo SJ, Savoie FH 3rd, Field LD, Ramsey JR, Hosemann CD. Tendinosis of the extensor carpi radialis brevis: an evaluation of three methods of operative treatment. J Shoulder Elbow Surg Am. 2006;15(6):721-727.
15. Almquist EE, Necking L, Bach AW. Epicondylar resection with anconeus transfer for chronic lateral epicondylitis. J Hand Surg Am. 1998;23(4):723-731.
16. Dunn JH, Kim JJ, Davis L, Nirschl RP. Ten- to 14-year follow-up of the Nirschl surgical technique for lateral epicondylitis. Am J Sports Med. 2008;36(2):261-266.
17. Rubenthaler F, Wiese M, Senge A, Keller L, Wittenberg RH. Long-term follow-up of open and endoscopic Hohmann procedures for lateral epicondylitis. Arthroscopy. 2005;21(6):684-690.
18. Lattermann C, Romeo AA, Anbari A, et al. Arthroscopic debridement of the extensor carpi radialis brevis for the treatment of recalcitrant lateral epicondylitis. J Shoulder Elbow Surg. 2010;19(5):651-656.
19. Rose NE, Forman SK, Dellon AL. Denervation of the lateral epicondyle for treatment of chronic lateral epicondylitis. J Hand Surg Am. 2013;38(2):344-349.
20. Koh JS, Mohan PC, Howe TS, et al. Fasciotomy and surgical tenotomy for recalcitrant lateral elbow tendonopathy: early clinical experience with a novel device for minimally invasive percutaneous microresection. Am J Sports Med. 2013;41(3):636-644.
21. Nirschl RP, Ashman ES. Elbow tendonopathy: tennis elbow. Clin Sports Med. 2003;22(4):813-836.
22. Dellon AL, Kim J, Ducic I. Painful neuroma of the posterior cutaneous nerve of the forearm after surgery for lateral humeral epicondylitis. J Hand Surg Am. 2004;29(3):387-390.
23. Cummins CA. Lateral epicondylitis: in-vivo assessment of arthroscopic debridement and correlation with patient outcomes. Am J Sports Med. 2006;34(9):1486-1491.
24. Meknas K, Odden-Miland A, Mercer JB, Castillejo M, Johansen O. Radiofrequency microtenotomy: a promising method for treatment of recalcitrant lateral epicondylitis. Am J Sports Med. 2008;36(10):1960-1965.
25. Takahashi N, Tasto JP, Locke J, et al. The use of radiofrequency (RF) for the treatment of chronic tendinosis. Paper presented at: 6th Biennial Congress of the International Society of Arthroscopy, Knee Surgery, and Orthopaedic Sports Medicine Congress; May 2007; Florence, Italy. Abstract 1433.
26. Takahashi N, Tasto JP, Ritter M, et al. Pain relief through an antinociceptive effect after radiofrequency application. Am J Sports Med. 2007;35(5):805-810.
27. Ochiai N, Tasto JP, Ohtori S, Takahashi N, Moriya H, Amiel D. Nerve regeneration after radiofrequency ablation. Am J Sports Med. 2007;35(11):1940-1944.
ABIM continues to lighten recertification load
AGA applauds the announcement by the American Board of Internal Medicine that it is extending the suspension of the practice improvement, patient safety, and patient voice requirements until at least December 2018.
Throughout 2015, AGA has pushed ABIM to reconsider the burdensome recertification process. Gastroenterologists need a recertification system that fosters active learning, not high-stakes testing.
Our campaign continues – AGA is communicating with other subspecialty societies to work together to secure the best approach to MOC consistent with the principles we previously published:
• MOC needs to be simpler, less intrusive, and less expensive.
• We support ending the high‐stakes, every-10‐year exam.
• We do not support closed‐book assessments as they do not represent the current realities of medicine in the digital age.
• We support the principles of lifelong learning as evidenced by ongoing CME activities, rather than lifelong testing.
• We support the concept that, for the many diplomates who specialize within certain areas of gastroenterology and hepatology, MOC should not need to include high‐stakes assessments of areas where the diplomate may not practice.
We hear you that MOC is a burden and we will continue to push for the principles of individualization, specialization, and dropping the high-stakes exam – sooner rather than later.
Practicalities for those up for recertification
While we advocate for a new MOC system, there are requirements that still stand. Make sure you are up to date before the end of 2015:
• If you need to complete MOC points by Dec. 31, 2015, AGA has activities you can complete to earn those points. Please visit the MOC section of the AGA website to see how we can help you.
• Each board certified physician’s requirements are slightly different based on the year of your certification or most recent recertification. For details specific to you, we urge you to log in to your ABIM Physician Portal.
For more information, read our paper, The Gastroenterologist-accountable Professionalism in Practice Pathway, and consensus principles for reform that AGA developed with ACG, ASGE, AASLD, ANMS and NASPGHAN.
AGA applauds the announcement by the American Board of Internal Medicine that it is extending the suspension of the practice improvement, patient safety, and patient voice requirements until at least December 2018.
Throughout 2015, AGA has pushed ABIM to reconsider the burdensome recertification process. Gastroenterologists need a recertification system that fosters active learning, not high-stakes testing.
Our campaign continues – AGA is communicating with other subspecialty societies to work together to secure the best approach to MOC consistent with the principles we previously published:
• MOC needs to be simpler, less intrusive, and less expensive.
• We support ending the high‐stakes, every-10‐year exam.
• We do not support closed‐book assessments as they do not represent the current realities of medicine in the digital age.
• We support the principles of lifelong learning as evidenced by ongoing CME activities, rather than lifelong testing.
• We support the concept that, for the many diplomates who specialize within certain areas of gastroenterology and hepatology, MOC should not need to include high‐stakes assessments of areas where the diplomate may not practice.
We hear you that MOC is a burden and we will continue to push for the principles of individualization, specialization, and dropping the high-stakes exam – sooner rather than later.
Practicalities for those up for recertification
While we advocate for a new MOC system, there are requirements that still stand. Make sure you are up to date before the end of 2015:
• If you need to complete MOC points by Dec. 31, 2015, AGA has activities you can complete to earn those points. Please visit the MOC section of the AGA website to see how we can help you.
• Each board certified physician’s requirements are slightly different based on the year of your certification or most recent recertification. For details specific to you, we urge you to log in to your ABIM Physician Portal.
For more information, read our paper, The Gastroenterologist-accountable Professionalism in Practice Pathway, and consensus principles for reform that AGA developed with ACG, ASGE, AASLD, ANMS and NASPGHAN.
AGA applauds the announcement by the American Board of Internal Medicine that it is extending the suspension of the practice improvement, patient safety, and patient voice requirements until at least December 2018.
Throughout 2015, AGA has pushed ABIM to reconsider the burdensome recertification process. Gastroenterologists need a recertification system that fosters active learning, not high-stakes testing.
Our campaign continues – AGA is communicating with other subspecialty societies to work together to secure the best approach to MOC consistent with the principles we previously published:
• MOC needs to be simpler, less intrusive, and less expensive.
• We support ending the high‐stakes, every-10‐year exam.
• We do not support closed‐book assessments as they do not represent the current realities of medicine in the digital age.
• We support the principles of lifelong learning as evidenced by ongoing CME activities, rather than lifelong testing.
• We support the concept that, for the many diplomates who specialize within certain areas of gastroenterology and hepatology, MOC should not need to include high‐stakes assessments of areas where the diplomate may not practice.
We hear you that MOC is a burden and we will continue to push for the principles of individualization, specialization, and dropping the high-stakes exam – sooner rather than later.
Practicalities for those up for recertification
While we advocate for a new MOC system, there are requirements that still stand. Make sure you are up to date before the end of 2015:
• If you need to complete MOC points by Dec. 31, 2015, AGA has activities you can complete to earn those points. Please visit the MOC section of the AGA website to see how we can help you.
• Each board certified physician’s requirements are slightly different based on the year of your certification or most recent recertification. For details specific to you, we urge you to log in to your ABIM Physician Portal.
For more information, read our paper, The Gastroenterologist-accountable Professionalism in Practice Pathway, and consensus principles for reform that AGA developed with ACG, ASGE, AASLD, ANMS and NASPGHAN.
Stewart Tepper, MD
The video associated with this article is no longer available on this site. Please view all of our videos on the MDedge YouTube channel
The video associated with this article is no longer available on this site. Please view all of our videos on the MDedge YouTube channel
The video associated with this article is no longer available on this site. Please view all of our videos on the MDedge YouTube channel
A girl refuses to eat solid food because she is afraid of choking
CASE Refusing solid food
Ms. B, age 11, is admitted to a pediatric medical inpatient unit for unintentional weight loss of 14 lb (15% total body weight) over the past month. She reports having 2 traumatic episodes last month: choking on a piece of cheese and having a swab specimen taken for a rapid strep test, which required several people to restrain her (the test was positive). Since then, she has refused to ingest solids, despite hunger and a desire to eat.
Ms. B reports diffuse abdominal pain merely “at the sight of food” and a fear of swallowing solids. She denies difficulty or pain upon swallowing, nausea, vomiting, or any change in bowel habits.
Her mother reports that, on the rare occasion that Ms. B has attempted to eat solid food, she spent as long as an hour cutting it into small pieces before bringing it to her mouth—after which she put the food down without eating. Her mother also witnessed Ms. B holding food in her mouth for “a very long time,” then spitting it out.
Ms. B says she is distressed about the weight loss and recognizes that her fear of solid food is excessive.
What would your diagnosis of Ms. B’s problem be?
a) anorexia nervosa
b) avoidant/restrictive food intake disorder (ARFID)
c) specific phobia (swallowing solids or choking)
d) generalized anxiety disorder (GAD)
The authors’ observations
DSM-5 describes a new eating disorder called ARFID, which replaces the DSM-IV-TR diagnosis of feeding disorder of infancy or early childhood</keyword>. DSM-5 diagnostic criteria define ARFID as:
An eating or feeding disturbance (eg, avoidance based on the sensory characteristics of food…) as manifested by persistent failure to meet appropriate nutritional and/or energy needs associated with at least one of the following: 1. Significant weight loss (or failure to achieve expected weight gain or faltering growth in children). 2. Significant nutritional deficiency. 3. Dependence on enteral feeding or oral nutritional supplements. 4. Marked interference with psychosocial functioning.1
DSM-5 also specifies that the disorder cannot be caused by lack of available food or traditional cultural practices; cannot coexist with anorexia or bulimia nervosa; and is not attributable to a concurrent medical or psychiatric disorder.
Because it is a newly defined diagnosis, the epidemiology of ARFID is unclear. Patients with ARFID have a wide variety of eating symptoms that do not meet diagnostic criteria for anorexia or bulimia nervosa. One study found that, among a cohort of mostly adolescent patients who presented for evaluation of an eating disorder, 14% met diagnostic criteria for ARFID.2 Another retrospective case-control study found a similar prevalence among patients age 8 to 18 (13.8% of 712 patients).3 Because of the variety of maladaptive feeding behaviors seen in ARFID, there is little evidence that pharmacotherapy is effective.4
HISTORY Premature birth
Ms. B’s medical history states that she is twin A of a premature birth at 26 weeks (birth weight, 1,060 g), with a 90-day neonatal intensive care unit hospitalization, during which she required supplemental oxygen and nasogastric tube feeding. She has mild cerebral palsy, and had motor delay of walking at 2.5 years old. Currently, she has no motor difficulties.
Ms. B does not have a psychiatric history and does not take medications. Her mother has a history of major depressive disorder that is well controlled with an unspecified selective serotonin reuptake inhibitor. Ms. B’s maternal uncle has poorly controlled schizophrenia.
During Ms. B’s 6-day hospitalization, her mental status exams are unremarkable. She is shy but cooperative and open. Her mood ranges from “sad” and “nervous” on admission to “fine” with mood-congruent affect. She denies suicidal or homicidal thoughts and hallucinations, and demonstrates good insight and judgment. All laboratory values are within normal limits except for mild hypophosphatemia (3.7 mg/dL) and mild hyperalbuminemia (4.9 g/dL) on admission, which may have been related to her nutritional status.
DIAGNOSIS Not solely psychiatric
The psychiatric differential diagnosis includes:
- ARFID
- specific phobia of swallowing solids or choking (pseudodysphagia)
- GAD
- unspecified feeding disorder.
Ms. B meets diagnostic criteria for ARFID, particularly that of profound acute weight loss due to restrictive eating behaviors. Her presentation also is similar to that of a specific phobia, namely profound anxiety upon even the thought of solid food (phobic stimulus) and recognition that her fear is excessive. However, she fails to meet diagnostic criteria for phobia in children, which require duration of at least 6 months. GAD also is less likely because she has had symptoms for 1 month (also requires 6-month duration) and her anxiety is limited to feeding behaviors.
The treatment team starts exposure therapy, encouraging Ms. B to begin taking small bites of textured foods, such as oatmeal.
On hospital Day 5, barium esophagogram reveals extrinsic compression of the esophagus. To identify the precise cause of compression, chest magnetic resonance angiogram reveals that Ms. B has a right-sided aortic arch with an aberrant left subclavian artery at T4 that, with the ligamentum arteriosum and left pulmonary artery, form a vascular ring impinging around the esophagus.
The authors’ observations
Dysphagia lusoria, coined in 1789 from the root for “natural abnormality,”5 is caused by an aberrant right subclavian artery, which persists because of abnormal involution of the right fourth aortic arch during embryogenesis6 (Figure 1). The condition is diagnosed incidentally in most affected adults, who are asymptomatic throughout life and do not require operative management.6
Symptoms of dysphagia lusoria can include dysphagia and recurrent aspiration if significant esophageal impingement is present.7 It is thought that patients tend to show more symptoms as they age because of sclerosis, aneurysm, or atherosclerosis of the impinging vessel.7 Cough and dyspnea caused by impingement of the trachea also have been reported, and might be more common in children because of tracheal flexibility.5
This case illustrates the complex interrelationship of physical and psychiatric conditions. After the treatment team discovered a physical cause of Ms. B’s symptoms, the initial psychiatric diagnosis became problematic, but remained critically important for long-term treatment of her comorbid eating phobia.
TREATMENT Therapy, surgery
The treatment team is faced with the question of whether dysphagia lusoria fully accounts for Ms. B’s status. The anatomic anomaly might explain the initial choking incident if the food particle was lodged at the site of impingement, but does not account for development of a severe acute eating disorder and subsequent malnutrition.
Because of the presence of dysphagia lusoria, the team concludes that Ms. B does not meet diagnostic criteria for ARFID. She is given a diagnosis of unspecified eating disorder.
Ms. B is discharged and referred to a pediatric cardiothoracic surgeon for operative consult of symptomatic dysphagia lusoria. By discharge, she is successfully eating yogurt with fruit chunks, which she had rejected earlier. She also expresses optimism about eating cake at her birthday party, scheduled for 2 weeks after discharge.
The treatment team strongly recommends outpatient psychiatric follow-up to manage Ms. B’s unspecified eating disorder with cognitive-behavioral therapy and exposure and response prevention, which helped to mildly decrease her anxiety during the hospital stay.
Ms. B’s surgeon deems the case severe enough to warrant surgery. Surgery for dysphagia lusoria can be indicated to prevent progression of symptoms into adulthood8; options include division of the ligamentum arteriosum to loosen the vascular ring and re-implantation of the aberrant subclavian artery.9,10 (On the other hand, dietary modification might be therapeutic in patients whose symptoms are mild.5)
The surgeon elects to divide the ligamentum arteriosum to relieve the pressure of the vascular ring on the esophagus. Intraoperatively, he discovers that Ms. B has a mildly patent ductus arteriosus (PDA) (Figure 2), which is more common in premature infants than in full-term births. The PDA is clipped on both sides and divided. This immediately causes the vascular ring created by the aberrant left subclavian artery, right-sided aorta, and PDA to spring open, releasing pressure on the esophagus.
The aberrant left subclavian artery emerges from an abnormal bulging of aorta, known as Kommerell’s diverticulum. After the vascular ring is released, the diverticulum is not observed to impinge on the esophagus; however, as a preventive measure, the surgeon sutures it to the anterior spinous ligament. This will prevent the diverticulum from enlarging and impinging on the esophagus as Ms. B grows to adulthood.
The surgery is completed without complications. Ms. B tolerates the procedure well.
Ten days after surgery, Ms. B is recovering well. She and her mother report satisfaction with the procedure to release the vascular ring. She discontinues her pain medication after 2 days and slowly begins reintroducing solid foods. Her fear of dysphagia and choking rapidly diminish.
Bottom Line
The diagnosis and management of eating disorders, including avoidant/restrictive food intake disorder and choking phobia, can be challenging. Furthermore, if an anatomical or organic anomaly is found, it is important to question how a patient’s eating disorder can be managed best through interdisciplinary collaboration between medical and behavioral specialties.
Related Resources
• Bryant-Waugh R. Feeding and eating disorders in children. Curr Opin Psychiatry. 2013;26(6):537-542.
• Norris ML, Robinson A, Obeid N, et al. Exploring avoidant/restrictive food intake disorder in eating disordered patients: a descriptive study. Int J Eat Disord. 2014;47(5):495-499.
Disclosures
The authors report no financial relationship with any company whose products are mentioned in this article or with manufacturers of competing products.
1. Diagnostic and statistical manual of mental disorders, fifth edition. Washington, DC: American Psychiatric Association; 2013.
2. Ornstein RM, Rosen DS, Mammel KA, et al. Distribution of eating disorders in children and adolescents using the proposed DSM-5 criteria for feeding and eating disorders. J Adolesc Health. 2013;53(2):303-305.
3. Fisher MM, Rosen DS, Ornstein RM, et al. Characteristics of avoidant/restrictive food intake disorder in children and adolescents: a “new disorder” in DSM-5. J Adolesc Health. 2014;55(1):49-52.
4. Kelly NR, Shank LM, Bakalar JL, et al. Pediatric feeding and eating disorders: current state of diagnosis and treatment. Curr Psychiatry Rep. 2014;16(5):446.
5. Janssen M, Baggen MG, Veen HF, et al. Dysphagia lusoria: clinical aspects, manometric findings, diagnosis, and therapy. Am J Gastroenterol. 2000;95(6):1411-1416.
6. Abraham V, Mathew A, Cherian V, et al. Aberrant subclavian artery: anatomical curiosity or clinical entity. Int J Surg. 2009;7(2):106-109.
7. Calleja F, Eguaras M, Montero J, et al. Aberrant right subclavian artery associated with common carotid trunk. A rare cause of vascular ring. Eur J Cardiothorac Surg. 1990;4(10):568-570.
8. Jalaie H, Grommes J, Sailer A, et al. Treatment of symptomatic aberrant subclavian arteries. Eur J Vasc Endovasc Surg. 2014;48(5):521-526.
9. Gross RE. Surgical treatment for dysphagia lusoria. Ann Surg. 1946;124:532-534.
10. Morrris CD, Kanter KR, Miller JI Jr. Late-onset dysphagia lusoria. Ann Thorac Surg. 2001;71(2):710-712.
CASE Refusing solid food
Ms. B, age 11, is admitted to a pediatric medical inpatient unit for unintentional weight loss of 14 lb (15% total body weight) over the past month. She reports having 2 traumatic episodes last month: choking on a piece of cheese and having a swab specimen taken for a rapid strep test, which required several people to restrain her (the test was positive). Since then, she has refused to ingest solids, despite hunger and a desire to eat.
Ms. B reports diffuse abdominal pain merely “at the sight of food” and a fear of swallowing solids. She denies difficulty or pain upon swallowing, nausea, vomiting, or any change in bowel habits.
Her mother reports that, on the rare occasion that Ms. B has attempted to eat solid food, she spent as long as an hour cutting it into small pieces before bringing it to her mouth—after which she put the food down without eating. Her mother also witnessed Ms. B holding food in her mouth for “a very long time,” then spitting it out.
Ms. B says she is distressed about the weight loss and recognizes that her fear of solid food is excessive.
What would your diagnosis of Ms. B’s problem be?
a) anorexia nervosa
b) avoidant/restrictive food intake disorder (ARFID)
c) specific phobia (swallowing solids or choking)
d) generalized anxiety disorder (GAD)
The authors’ observations
DSM-5 describes a new eating disorder called ARFID, which replaces the DSM-IV-TR diagnosis of feeding disorder of infancy or early childhood</keyword>. DSM-5 diagnostic criteria define ARFID as:
An eating or feeding disturbance (eg, avoidance based on the sensory characteristics of food…) as manifested by persistent failure to meet appropriate nutritional and/or energy needs associated with at least one of the following: 1. Significant weight loss (or failure to achieve expected weight gain or faltering growth in children). 2. Significant nutritional deficiency. 3. Dependence on enteral feeding or oral nutritional supplements. 4. Marked interference with psychosocial functioning.1
DSM-5 also specifies that the disorder cannot be caused by lack of available food or traditional cultural practices; cannot coexist with anorexia or bulimia nervosa; and is not attributable to a concurrent medical or psychiatric disorder.
Because it is a newly defined diagnosis, the epidemiology of ARFID is unclear. Patients with ARFID have a wide variety of eating symptoms that do not meet diagnostic criteria for anorexia or bulimia nervosa. One study found that, among a cohort of mostly adolescent patients who presented for evaluation of an eating disorder, 14% met diagnostic criteria for ARFID.2 Another retrospective case-control study found a similar prevalence among patients age 8 to 18 (13.8% of 712 patients).3 Because of the variety of maladaptive feeding behaviors seen in ARFID, there is little evidence that pharmacotherapy is effective.4
HISTORY Premature birth
Ms. B’s medical history states that she is twin A of a premature birth at 26 weeks (birth weight, 1,060 g), with a 90-day neonatal intensive care unit hospitalization, during which she required supplemental oxygen and nasogastric tube feeding. She has mild cerebral palsy, and had motor delay of walking at 2.5 years old. Currently, she has no motor difficulties.
Ms. B does not have a psychiatric history and does not take medications. Her mother has a history of major depressive disorder that is well controlled with an unspecified selective serotonin reuptake inhibitor. Ms. B’s maternal uncle has poorly controlled schizophrenia.
During Ms. B’s 6-day hospitalization, her mental status exams are unremarkable. She is shy but cooperative and open. Her mood ranges from “sad” and “nervous” on admission to “fine” with mood-congruent affect. She denies suicidal or homicidal thoughts and hallucinations, and demonstrates good insight and judgment. All laboratory values are within normal limits except for mild hypophosphatemia (3.7 mg/dL) and mild hyperalbuminemia (4.9 g/dL) on admission, which may have been related to her nutritional status.
DIAGNOSIS Not solely psychiatric
The psychiatric differential diagnosis includes:
- ARFID
- specific phobia of swallowing solids or choking (pseudodysphagia)
- GAD
- unspecified feeding disorder.
Ms. B meets diagnostic criteria for ARFID, particularly that of profound acute weight loss due to restrictive eating behaviors. Her presentation also is similar to that of a specific phobia, namely profound anxiety upon even the thought of solid food (phobic stimulus) and recognition that her fear is excessive. However, she fails to meet diagnostic criteria for phobia in children, which require duration of at least 6 months. GAD also is less likely because she has had symptoms for 1 month (also requires 6-month duration) and her anxiety is limited to feeding behaviors.
The treatment team starts exposure therapy, encouraging Ms. B to begin taking small bites of textured foods, such as oatmeal.
On hospital Day 5, barium esophagogram reveals extrinsic compression of the esophagus. To identify the precise cause of compression, chest magnetic resonance angiogram reveals that Ms. B has a right-sided aortic arch with an aberrant left subclavian artery at T4 that, with the ligamentum arteriosum and left pulmonary artery, form a vascular ring impinging around the esophagus.
The authors’ observations
Dysphagia lusoria, coined in 1789 from the root for “natural abnormality,”5 is caused by an aberrant right subclavian artery, which persists because of abnormal involution of the right fourth aortic arch during embryogenesis6 (Figure 1). The condition is diagnosed incidentally in most affected adults, who are asymptomatic throughout life and do not require operative management.6
Symptoms of dysphagia lusoria can include dysphagia and recurrent aspiration if significant esophageal impingement is present.7 It is thought that patients tend to show more symptoms as they age because of sclerosis, aneurysm, or atherosclerosis of the impinging vessel.7 Cough and dyspnea caused by impingement of the trachea also have been reported, and might be more common in children because of tracheal flexibility.5
This case illustrates the complex interrelationship of physical and psychiatric conditions. After the treatment team discovered a physical cause of Ms. B’s symptoms, the initial psychiatric diagnosis became problematic, but remained critically important for long-term treatment of her comorbid eating phobia.
TREATMENT Therapy, surgery
The treatment team is faced with the question of whether dysphagia lusoria fully accounts for Ms. B’s status. The anatomic anomaly might explain the initial choking incident if the food particle was lodged at the site of impingement, but does not account for development of a severe acute eating disorder and subsequent malnutrition.
Because of the presence of dysphagia lusoria, the team concludes that Ms. B does not meet diagnostic criteria for ARFID. She is given a diagnosis of unspecified eating disorder.
Ms. B is discharged and referred to a pediatric cardiothoracic surgeon for operative consult of symptomatic dysphagia lusoria. By discharge, she is successfully eating yogurt with fruit chunks, which she had rejected earlier. She also expresses optimism about eating cake at her birthday party, scheduled for 2 weeks after discharge.
The treatment team strongly recommends outpatient psychiatric follow-up to manage Ms. B’s unspecified eating disorder with cognitive-behavioral therapy and exposure and response prevention, which helped to mildly decrease her anxiety during the hospital stay.
Ms. B’s surgeon deems the case severe enough to warrant surgery. Surgery for dysphagia lusoria can be indicated to prevent progression of symptoms into adulthood8; options include division of the ligamentum arteriosum to loosen the vascular ring and re-implantation of the aberrant subclavian artery.9,10 (On the other hand, dietary modification might be therapeutic in patients whose symptoms are mild.5)
The surgeon elects to divide the ligamentum arteriosum to relieve the pressure of the vascular ring on the esophagus. Intraoperatively, he discovers that Ms. B has a mildly patent ductus arteriosus (PDA) (Figure 2), which is more common in premature infants than in full-term births. The PDA is clipped on both sides and divided. This immediately causes the vascular ring created by the aberrant left subclavian artery, right-sided aorta, and PDA to spring open, releasing pressure on the esophagus.
The aberrant left subclavian artery emerges from an abnormal bulging of aorta, known as Kommerell’s diverticulum. After the vascular ring is released, the diverticulum is not observed to impinge on the esophagus; however, as a preventive measure, the surgeon sutures it to the anterior spinous ligament. This will prevent the diverticulum from enlarging and impinging on the esophagus as Ms. B grows to adulthood.
The surgery is completed without complications. Ms. B tolerates the procedure well.
Ten days after surgery, Ms. B is recovering well. She and her mother report satisfaction with the procedure to release the vascular ring. She discontinues her pain medication after 2 days and slowly begins reintroducing solid foods. Her fear of dysphagia and choking rapidly diminish.
Bottom Line
The diagnosis and management of eating disorders, including avoidant/restrictive food intake disorder and choking phobia, can be challenging. Furthermore, if an anatomical or organic anomaly is found, it is important to question how a patient’s eating disorder can be managed best through interdisciplinary collaboration between medical and behavioral specialties.
Related Resources
• Bryant-Waugh R. Feeding and eating disorders in children. Curr Opin Psychiatry. 2013;26(6):537-542.
• Norris ML, Robinson A, Obeid N, et al. Exploring avoidant/restrictive food intake disorder in eating disordered patients: a descriptive study. Int J Eat Disord. 2014;47(5):495-499.
Disclosures
The authors report no financial relationship with any company whose products are mentioned in this article or with manufacturers of competing products.
CASE Refusing solid food
Ms. B, age 11, is admitted to a pediatric medical inpatient unit for unintentional weight loss of 14 lb (15% total body weight) over the past month. She reports having 2 traumatic episodes last month: choking on a piece of cheese and having a swab specimen taken for a rapid strep test, which required several people to restrain her (the test was positive). Since then, she has refused to ingest solids, despite hunger and a desire to eat.
Ms. B reports diffuse abdominal pain merely “at the sight of food” and a fear of swallowing solids. She denies difficulty or pain upon swallowing, nausea, vomiting, or any change in bowel habits.
Her mother reports that, on the rare occasion that Ms. B has attempted to eat solid food, she spent as long as an hour cutting it into small pieces before bringing it to her mouth—after which she put the food down without eating. Her mother also witnessed Ms. B holding food in her mouth for “a very long time,” then spitting it out.
Ms. B says she is distressed about the weight loss and recognizes that her fear of solid food is excessive.
What would your diagnosis of Ms. B’s problem be?
a) anorexia nervosa
b) avoidant/restrictive food intake disorder (ARFID)
c) specific phobia (swallowing solids or choking)
d) generalized anxiety disorder (GAD)
The authors’ observations
DSM-5 describes a new eating disorder called ARFID, which replaces the DSM-IV-TR diagnosis of feeding disorder of infancy or early childhood</keyword>. DSM-5 diagnostic criteria define ARFID as:
An eating or feeding disturbance (eg, avoidance based on the sensory characteristics of food…) as manifested by persistent failure to meet appropriate nutritional and/or energy needs associated with at least one of the following: 1. Significant weight loss (or failure to achieve expected weight gain or faltering growth in children). 2. Significant nutritional deficiency. 3. Dependence on enteral feeding or oral nutritional supplements. 4. Marked interference with psychosocial functioning.1
DSM-5 also specifies that the disorder cannot be caused by lack of available food or traditional cultural practices; cannot coexist with anorexia or bulimia nervosa; and is not attributable to a concurrent medical or psychiatric disorder.
Because it is a newly defined diagnosis, the epidemiology of ARFID is unclear. Patients with ARFID have a wide variety of eating symptoms that do not meet diagnostic criteria for anorexia or bulimia nervosa. One study found that, among a cohort of mostly adolescent patients who presented for evaluation of an eating disorder, 14% met diagnostic criteria for ARFID.2 Another retrospective case-control study found a similar prevalence among patients age 8 to 18 (13.8% of 712 patients).3 Because of the variety of maladaptive feeding behaviors seen in ARFID, there is little evidence that pharmacotherapy is effective.4
HISTORY Premature birth
Ms. B’s medical history states that she is twin A of a premature birth at 26 weeks (birth weight, 1,060 g), with a 90-day neonatal intensive care unit hospitalization, during which she required supplemental oxygen and nasogastric tube feeding. She has mild cerebral palsy, and had motor delay of walking at 2.5 years old. Currently, she has no motor difficulties.
Ms. B does not have a psychiatric history and does not take medications. Her mother has a history of major depressive disorder that is well controlled with an unspecified selective serotonin reuptake inhibitor. Ms. B’s maternal uncle has poorly controlled schizophrenia.
During Ms. B’s 6-day hospitalization, her mental status exams are unremarkable. She is shy but cooperative and open. Her mood ranges from “sad” and “nervous” on admission to “fine” with mood-congruent affect. She denies suicidal or homicidal thoughts and hallucinations, and demonstrates good insight and judgment. All laboratory values are within normal limits except for mild hypophosphatemia (3.7 mg/dL) and mild hyperalbuminemia (4.9 g/dL) on admission, which may have been related to her nutritional status.
DIAGNOSIS Not solely psychiatric
The psychiatric differential diagnosis includes:
- ARFID
- specific phobia of swallowing solids or choking (pseudodysphagia)
- GAD
- unspecified feeding disorder.
Ms. B meets diagnostic criteria for ARFID, particularly that of profound acute weight loss due to restrictive eating behaviors. Her presentation also is similar to that of a specific phobia, namely profound anxiety upon even the thought of solid food (phobic stimulus) and recognition that her fear is excessive. However, she fails to meet diagnostic criteria for phobia in children, which require duration of at least 6 months. GAD also is less likely because she has had symptoms for 1 month (also requires 6-month duration) and her anxiety is limited to feeding behaviors.
The treatment team starts exposure therapy, encouraging Ms. B to begin taking small bites of textured foods, such as oatmeal.
On hospital Day 5, barium esophagogram reveals extrinsic compression of the esophagus. To identify the precise cause of compression, chest magnetic resonance angiogram reveals that Ms. B has a right-sided aortic arch with an aberrant left subclavian artery at T4 that, with the ligamentum arteriosum and left pulmonary artery, form a vascular ring impinging around the esophagus.
The authors’ observations
Dysphagia lusoria, coined in 1789 from the root for “natural abnormality,”5 is caused by an aberrant right subclavian artery, which persists because of abnormal involution of the right fourth aortic arch during embryogenesis6 (Figure 1). The condition is diagnosed incidentally in most affected adults, who are asymptomatic throughout life and do not require operative management.6
Symptoms of dysphagia lusoria can include dysphagia and recurrent aspiration if significant esophageal impingement is present.7 It is thought that patients tend to show more symptoms as they age because of sclerosis, aneurysm, or atherosclerosis of the impinging vessel.7 Cough and dyspnea caused by impingement of the trachea also have been reported, and might be more common in children because of tracheal flexibility.5
This case illustrates the complex interrelationship of physical and psychiatric conditions. After the treatment team discovered a physical cause of Ms. B’s symptoms, the initial psychiatric diagnosis became problematic, but remained critically important for long-term treatment of her comorbid eating phobia.
TREATMENT Therapy, surgery
The treatment team is faced with the question of whether dysphagia lusoria fully accounts for Ms. B’s status. The anatomic anomaly might explain the initial choking incident if the food particle was lodged at the site of impingement, but does not account for development of a severe acute eating disorder and subsequent malnutrition.
Because of the presence of dysphagia lusoria, the team concludes that Ms. B does not meet diagnostic criteria for ARFID. She is given a diagnosis of unspecified eating disorder.
Ms. B is discharged and referred to a pediatric cardiothoracic surgeon for operative consult of symptomatic dysphagia lusoria. By discharge, she is successfully eating yogurt with fruit chunks, which she had rejected earlier. She also expresses optimism about eating cake at her birthday party, scheduled for 2 weeks after discharge.
The treatment team strongly recommends outpatient psychiatric follow-up to manage Ms. B’s unspecified eating disorder with cognitive-behavioral therapy and exposure and response prevention, which helped to mildly decrease her anxiety during the hospital stay.
Ms. B’s surgeon deems the case severe enough to warrant surgery. Surgery for dysphagia lusoria can be indicated to prevent progression of symptoms into adulthood8; options include division of the ligamentum arteriosum to loosen the vascular ring and re-implantation of the aberrant subclavian artery.9,10 (On the other hand, dietary modification might be therapeutic in patients whose symptoms are mild.5)
The surgeon elects to divide the ligamentum arteriosum to relieve the pressure of the vascular ring on the esophagus. Intraoperatively, he discovers that Ms. B has a mildly patent ductus arteriosus (PDA) (Figure 2), which is more common in premature infants than in full-term births. The PDA is clipped on both sides and divided. This immediately causes the vascular ring created by the aberrant left subclavian artery, right-sided aorta, and PDA to spring open, releasing pressure on the esophagus.
The aberrant left subclavian artery emerges from an abnormal bulging of aorta, known as Kommerell’s diverticulum. After the vascular ring is released, the diverticulum is not observed to impinge on the esophagus; however, as a preventive measure, the surgeon sutures it to the anterior spinous ligament. This will prevent the diverticulum from enlarging and impinging on the esophagus as Ms. B grows to adulthood.
The surgery is completed without complications. Ms. B tolerates the procedure well.
Ten days after surgery, Ms. B is recovering well. She and her mother report satisfaction with the procedure to release the vascular ring. She discontinues her pain medication after 2 days and slowly begins reintroducing solid foods. Her fear of dysphagia and choking rapidly diminish.
Bottom Line
The diagnosis and management of eating disorders, including avoidant/restrictive food intake disorder and choking phobia, can be challenging. Furthermore, if an anatomical or organic anomaly is found, it is important to question how a patient’s eating disorder can be managed best through interdisciplinary collaboration between medical and behavioral specialties.
Related Resources
• Bryant-Waugh R. Feeding and eating disorders in children. Curr Opin Psychiatry. 2013;26(6):537-542.
• Norris ML, Robinson A, Obeid N, et al. Exploring avoidant/restrictive food intake disorder in eating disordered patients: a descriptive study. Int J Eat Disord. 2014;47(5):495-499.
Disclosures
The authors report no financial relationship with any company whose products are mentioned in this article or with manufacturers of competing products.
1. Diagnostic and statistical manual of mental disorders, fifth edition. Washington, DC: American Psychiatric Association; 2013.
2. Ornstein RM, Rosen DS, Mammel KA, et al. Distribution of eating disorders in children and adolescents using the proposed DSM-5 criteria for feeding and eating disorders. J Adolesc Health. 2013;53(2):303-305.
3. Fisher MM, Rosen DS, Ornstein RM, et al. Characteristics of avoidant/restrictive food intake disorder in children and adolescents: a “new disorder” in DSM-5. J Adolesc Health. 2014;55(1):49-52.
4. Kelly NR, Shank LM, Bakalar JL, et al. Pediatric feeding and eating disorders: current state of diagnosis and treatment. Curr Psychiatry Rep. 2014;16(5):446.
5. Janssen M, Baggen MG, Veen HF, et al. Dysphagia lusoria: clinical aspects, manometric findings, diagnosis, and therapy. Am J Gastroenterol. 2000;95(6):1411-1416.
6. Abraham V, Mathew A, Cherian V, et al. Aberrant subclavian artery: anatomical curiosity or clinical entity. Int J Surg. 2009;7(2):106-109.
7. Calleja F, Eguaras M, Montero J, et al. Aberrant right subclavian artery associated with common carotid trunk. A rare cause of vascular ring. Eur J Cardiothorac Surg. 1990;4(10):568-570.
8. Jalaie H, Grommes J, Sailer A, et al. Treatment of symptomatic aberrant subclavian arteries. Eur J Vasc Endovasc Surg. 2014;48(5):521-526.
9. Gross RE. Surgical treatment for dysphagia lusoria. Ann Surg. 1946;124:532-534.
10. Morrris CD, Kanter KR, Miller JI Jr. Late-onset dysphagia lusoria. Ann Thorac Surg. 2001;71(2):710-712.
1. Diagnostic and statistical manual of mental disorders, fifth edition. Washington, DC: American Psychiatric Association; 2013.
2. Ornstein RM, Rosen DS, Mammel KA, et al. Distribution of eating disorders in children and adolescents using the proposed DSM-5 criteria for feeding and eating disorders. J Adolesc Health. 2013;53(2):303-305.
3. Fisher MM, Rosen DS, Ornstein RM, et al. Characteristics of avoidant/restrictive food intake disorder in children and adolescents: a “new disorder” in DSM-5. J Adolesc Health. 2014;55(1):49-52.
4. Kelly NR, Shank LM, Bakalar JL, et al. Pediatric feeding and eating disorders: current state of diagnosis and treatment. Curr Psychiatry Rep. 2014;16(5):446.
5. Janssen M, Baggen MG, Veen HF, et al. Dysphagia lusoria: clinical aspects, manometric findings, diagnosis, and therapy. Am J Gastroenterol. 2000;95(6):1411-1416.
6. Abraham V, Mathew A, Cherian V, et al. Aberrant subclavian artery: anatomical curiosity or clinical entity. Int J Surg. 2009;7(2):106-109.
7. Calleja F, Eguaras M, Montero J, et al. Aberrant right subclavian artery associated with common carotid trunk. A rare cause of vascular ring. Eur J Cardiothorac Surg. 1990;4(10):568-570.
8. Jalaie H, Grommes J, Sailer A, et al. Treatment of symptomatic aberrant subclavian arteries. Eur J Vasc Endovasc Surg. 2014;48(5):521-526.
9. Gross RE. Surgical treatment for dysphagia lusoria. Ann Surg. 1946;124:532-534.
10. Morrris CD, Kanter KR, Miller JI Jr. Late-onset dysphagia lusoria. Ann Thorac Surg. 2001;71(2):710-712.
Pigmented Villonodular Synovitis of the Hip: A Systematic Review
Pigmented villonodular synovitis (PVNS) is a rare monoarticular disorder that affects the joints, bursae, or tendon sheaths of 1.8 per million patients.1,2 PVNS is defined by exuberant proliferation of synovial villi and nodules. Although its etiology is unknown, PVNS behaves much as a neoplastic process does, with occasional chromosomal abnormalities, local tissue invasion, and the potential for malignant transformation.3,4 Radiographs show cystic erosions or joint space narrowing, and magnetic resonance imaging shows characteristic low-signal intensity (on T1- and T2-weighted sequences) because of high hemosiderin content. Biopsy remains the gold standard for diagnosis and reveals hemosiderin-laden macrophages, vascularized villi, mononuclear cell infiltration, and sporadic mitotic figures.5 Diffuse PVNS appears as a thickened synovium with matted villi and synovial folds; localized PVNS presents as a pedunculated, firm yellow nodule.6
PVNS has a predilection for large joints, most commonly the knee (up to 80% of cases) and the hip.1,2,7 Treatment strategies for knee PVNS have been well studied and, as an aggregate, show no superiority of arthroscopic or open techniques.8 The literature on hip PVNS is less abundant and more case-based, making it difficult to reach a consensus on effective treatment. Open synovectomy and arthroplasty have been the mainstays of treatment over the past 60 years, but the advent of hip arthroscopy has introduced a new treatment modality.1,9 As arthroscopic management becomes more readily available, it is important to understand and compare the effectiveness of synovectomy and arthroplasty.
We systematically reviewed the treatment modalities for PVNS of the hip to determine how synovectomy and arthroplasty compare with respect to efficacy and revision rates.
Methods
Search Strategy
We systematically reviewed the literature according to PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) guidelines using the PRISMA checklist.10 Searches were completed in July 2014 using the PubMed Medline database and the Cochrane Central Register of Clinical Trials. Keyword selection was designed to capture all level I to V evidence English-language studies that reported clinical and/or radiographic outcomes. This was accomplished with a keyword search of all available titles and manuscript abstracts: (pigmented [Title/Abstract] AND villonodular [Title/Abstract] AND synovitis [Title/Abstract]) AND (hip [Title/Abstract]) AND (English [lang])). Abstracts from the 75 resulting studies were reviewed for exclusion criteria, which consisted of any cadaveric, biomechanical, histologic, and/or kinematic results, as well as a lack of any clinical and/or radiographic data (eg, review or technique articles). Studies were also excluded if they did not have clinical follow-up of at least 2 years. Studies not dedicated to hip PVNS specifically were not immediately excluded but were reviewed for outcomes data specific to the hip PVNS subpopulation. If a specific hip PVNS population could be distinguished from other patients, that study was included for review. If a study could not be deconstructed as such or was entirely devoted to one of our exclusion criteria, that study was excluded from our review. This initial search strategy yielded 16 studies.1,6,7,11-28
Bibliographical review of these 16 studies yielded several more for review. To ensure that no patients were counted twice, each study’s authors, data collection period, and ethnic population were reviewed and compared with those of the other studies. If there was any overlap in authorship, period, and place, only the study with the most relevant or comprehensive data was included. After accounting for all inclusion and exclusion criteria, we selected a total of 21 studies with 82 patients (86 hips) for inclusion (Figure 1).
Data Extraction
Details of study design, sample size, and patient demographics, including age, sex, and duration of symptoms, were recorded. Use of diagnostic biopsy, joint space narrowing on radiographs, treatment method, and use of radiation therapy were also abstracted. Some studies described multiple treatment methods. If those methods could not be differentiated into distinct outcomes groups, the study would have been excluded for lack of specific clinical data. Studies with sufficient data were deconstructed such that the patients from each treatment group were isolated.
Fewer than 5 studies reported physical examination findings, validated survey scores, and/or radiographic results. Therefore, the primary outcomes reported and compared between treatment groups were disease recurrence, clinical worsening defined as progressive pain or loss of function, and revision surgery. Revision surgery was subdivided into repeat synovectomy and eventual arthroplasty, arthrodesis, or revision arthroplasty. Time to revision surgery was also documented. Each study’s methodologic quality and bias were evaluated with the Modified Coleman Methodology Score (MCMS), described by Cowan and colleagues.29 MCMS is a 15-item instrument that has been used to assess randomized and nonrandomized patient trials.30,31 It has a scaled potential score ranging from 0 to 100, with scores from 85 through 100 indicating excellent, 70 through 84 good, 55 through 69 fair, and under 55 poor.
Statistical Analysis
We report our data as weighted means (SDs). A mean was calculated for each study reporting on a respective data point, and each mean was then weighted according to the sample size of that study. We multiplied each study’s individual mean by the number of patients enrolled in that study and divided the sum of all the studies’ weighted data points by the number of eligible patients in all relevant studies. The result is that the nonweighted means from studies with a smaller sample size did not carry as much weight as those from larger studies. We then compared 2 groups of patients: those who had only a synovectomy and those who had a combination of synovectomy and arthroplasty. The synovectomy-only group was also compared with a group that underwent total hip arthroplasty (THA) specifically (Figure 2). Groups were compared with Student t test (SPSS Version 18, IBM), and statistical significance was set at α = 0.05.
Results
Twenty-one studies (82 patients) were included in the final dataset (Table 1). Of these studies, 19 were retrospective case series (level IV evidence) in which the number of eligible hip PVNS patients ranged from 1 to 15. The other 2 studies were case reports (level V evidence). Mean (SD) MCMS was 25.0 (10.9).
Fifty-one patients (59.3%) were female. Mean (SD) age of all patients was 33.2 (12.6) years. Mean (SD) duration of symptoms was 4.2 (2.7) years. The right hip was affected in 59.5% of patients in whom laterality was documented. Sixty-eight patients (79.1%) had biopsy-proven PVNS; presence or absence of a biopsy was not documented for the other 18 patients.
Of the 82 patients in the study, 45 (54.9%) underwent synovectomy without arthroplasty. Staged radiation was used to augment the synovectomy in 2 of these 45 cases. One series in this group consisted of 15 cases of arthroscopic synovectomy.1 The 37 patients (45.1%) in the other treatment group had arthroplasty at time of synovectomy. These patients underwent 22 THAs, 8 cup arthroplasties, 2 metal-on-metal hip resurfacings, and 1 hemiarthroplasty. The remaining 4 patients were treated nonoperatively (3) or with primary arthrodesis (1).
Comparisons between the synovectomy-only and synovectomy-with-arthroplasty groups are listed in Table 2. Synovectomy patients were younger on average than arthroplasty patients, but the difference was not statistically significant (P = .28). Only 6 studies distinguished between local and diffuse PVNS histology, and the diffuse type was detected in 87.0%, with insufficient data to detect a difference between the synovectomy and arthroplasty groups. In studies with documented radiographic findings, 75.0% of patients had evidence of joint space narrowing, which was significantly (P = .03) more common in the arthroplasty group (96.7% vs 31.3%).
Mean (SD) clinical follow-up was 8.4 (5.9) years for all patients. A larger percentage of synovectomy-only patients experienced recurrence and worsened symptoms, but neither trend achieved statistical significance. The rate of eventual THA or arthrodesis after synovectomy alone was almost identical (P = .17) to the rate of revision THA in the synovectomy-with-arthroplasty group (26.2% vs 24.3%). Time to revision surgery, however, was significantly (P = .02) longer in the arthroplasty group. Two additional patients in the synovectomy-with-arthroplasty group underwent repeat synovectomy alone, but no patients in the synovectomy-only group underwent repeat synovectomy without arthroplasty.
One nonoperatively managed patient experienced symptom progression over the course of 10 years. The other 2 patients were stable after 2- and 4-year follow-up. The arthrodesis patient did not experience recurrence or have a revision operation in the 5 years after the index procedure.
Discussion
PVNS is a proliferative disorder of synovial tissue with a high risk of recurrence.15,32 Metastasis is extremely rare; there is only 1 case report of a fatality, which occurred within 42 months.12 Chiari and colleagues15 suggested that the PVNS recurrence rate is highest in the large joints. Therefore, in hip PVNS, early surgical resection is needed to limit articular destruction and the potential for recurrence. The primary treatment modalities are synovectomy alone and synovectomy with arthroplasty, which includes THA, cup arthroplasty, hip resurfacing, and hemiarthroplasty. According to our systematic review, about one-fourth of all patients in both treatment groups ultimately underwent revision surgery. Mean time to revision was significantly longer for synovectomy-with-arthroplasty patients (almost 12 years) than for synovectomy-only patients (6.5 years). One potential explanation is that arthroplasty component fixation may take longer to loosen than an inadequately synovectomized joint takes to recur. The synovectomy-only group did have a higher recurrence rate, though the difference was not statistically significant.
Open synovectomy is the most widely described technique for addressing hip PVNS. The precise pathophysiology of PVNS remains largely unknown, but most authors agree that aggressive débridement is required to halt its locally invasive course. Scott24 described the invasion of vascular foramina from synovium into bone and thought that radical synovectomy was essential to remove the stalks of these synovial villi. Furthermore, PVNS most commonly affects adults in the third through fifth decades of life,7 and many surgeons want to avoid prosthetic components (which may loosen over time) in this age group. Synovectomy, however, has persistently high recurrence rates, and, without removal of the femoral head and neck, it can be difficult to obtain adequate exposure for complete débridement. Although adjuvant external beam radiation has been used by some authors,17,19,33 its utility is unproven, and other authors have cautioned against unnecessary irradiation of reproductive organs.1,24,34
The high rates of bony involvement, joint destruction, and recurrence after synovectomy have prompted many surgeons to turn to arthroplasty. González Della Valle and colleagues18 theorized that joint space narrowing is more common in hip PVNS because of the poor distensibility of the hip capsule compared with that of the knee and other joints. In turn, bony lesions and arthritis present earlier in hip PVNS.14 Yoo and colleagues14 found a statistically significant increase in Harris Hip Scale (HHS) scores and a high rate of return to athletic activity after THA for PVNS. However, they also reported revisions for component loosening and osteolysis in 2 of 8 patients and periprosthetic osteolysis without loosening in another 2 patients. Vastel and colleagues16 similarly reported aseptic loosening of the acetabular component in half their patient cohort. No studies have determined which condition—PVNS recurrence or debris-related osteolysis—causes the accelerated loosening in this demographic.
Byrd and colleagues1 recently described use of hip arthroscopy in the treatment of PVNS. In a cohort of 13 patients, they found statistically significant improvements in HHS scores, no postoperative complications, and only 1 revision (THA 6 years after surgery). Although there is a prevailing perception that nodular (vs diffuse) PVNS is more appropriately treated with arthroscopic excision, no studies have provided data on this effect, and Byrd and colleagues1 in fact showed a trend of slightly better outcomes in diffuse cases than in nodular cases. The main challenges of hip arthroscopy are the steep learning curve and adequate exposure. Recent innovations include additional arthroscopic portals and enlarged T-capsulotomy, which may be contributing to decreased complication rates in hip arthroscopy in general.35
The limitations of this systematic review were largely imposed by the studies analyzed. The primary limitation was the relative paucity of clinical and radiographic data on hip PVNS. To our knowledge, studies on the treatment of hip PVNS have reported evidence levels no higher than IV. In addition, the studies we reviewed often had only 1 or 2 patient cases satisfying our inclusion criteria. For this reason, we included case reports, which further lowered the level of evidence of studies used. There were no consistently reported physical examination, survey, or radiographic findings that could be used to compare studies. All studies with sufficient data on hip PVNS treatment outcomes were rated poorly with the Modified Coleman Methodology Scoring system.29 Selection bias was minimized by the inclusive nature of studies with level I to V evidence, but this led to a study design bias in that most studies consisted of level IV evidence.
Conclusion
Although the hip PVNS literature is limited, our review provides insight into expected outcomes. No matter which surgery is to be performed, surgeons must counsel patients about the high revision rate. One in 4 patients ultimately undergoes a second surgery, which may be required within 6 or 7 years after synovectomy without arthroplasty. Further development and innovation in hip arthroscopy may transform the treatment of PVNS. We encourage other investigators to conduct prospective, comparative trials with higher evidence levels to assess the utility of arthroscopy and other treatment modalities.
1. Byrd JWT, Jones KS, Maiers GP. Two to 10 years’ follow-up of arthroscopic management of pigmented villonodular synovitis in the hip: a case series. Arthroscopy. 2013;29(11):1783-1787.
2. Myers BW, Masi AT. Pigmented villonodular synovitis and tenosynovitis: a clinical epidemiologic study of 166 cases and literature review. Medicine. 1980;59(3):223-238.
3. Sciot R, Rosai J, Dal Cin P, et al. Analysis of 35 cases of localized and diffuse tenosynovial giant cell tumor: a report from the Chromosomes and Morphology (CHAMP) study group. Mod Pathol. 1999;12(6):576-579.
4. Bertoni F, Unni KK, Beabout JW, Sim FH. Malignant giant cell tumor of the tendon sheaths and joints (malignant pigmented villonodular synovitis). Am J Surg Pathol. 1997;21(2):153-163.
5. Mankin H, Trahan C, Hornicek F. Pigmented villonodular synovitis of joints. J Surg Oncol. 2011;103(5):386-389.
6. Martin RC, Osborne DL, Edwards MJ, Wrightson W, McMasters KM. Giant cell tumor of tendon sheath, tenosynovial giant cell tumor, and pigmented villonodular synovitis: defining the presentation, surgical therapy and recurrence. Oncol Rep. 2000;7(2):413-419.
7. Danzig LA, Gershuni DH, Resnick D. Diagnosis and treatment of diffuse pigmented villonodular synovitis of the hip. Clin Orthop Relat Res. 1982;(168):42-47.
8. Aurégan JC, Klouche S, Bohu Y, Lefèvre N, Herman S, Hardy P. Treatment of pigmented villonodular synovitis of the knee. Arthroscopy. 2014;30(10):1327-1341.
9. Gondolph-Zink B, Puhl W, Noack W. Semiarthroscopic synovectomy of the hip. Int Orthop. 1988;12(1):31-35.
10. Moher D, Liberati A, Tetzlaff J, Altman DG; PRISMA Group. Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. J Clin Epidemiol. 2009;62(10):1006-1012.
11. Shoji T, Yasunaga Y, Yamasaki T, et al. Transtrochanteric rotational osteotomy combined with intra-articular procedures for pigmented villonodular synovitis of the hip. J Orthop Sci. 2015;20(5):943-950.
12. Li LM, Jeffery J. Exceptionally aggressive pigmented villonodular synovitis of the hip unresponsive to radiotherapy. J Bone Joint Surg Br. 2011;93(7):995-997.
13. Hoberg M, Amstutz HC. Metal-on-metal hip resurfacing in patients with pigmented villonodular synovitis: a report of two cases. Orthopedics. 2010;33(1):50-53.
14. Yoo JJ, Kwon YS, Koo KH, Yoon KS, Min BW, Kim HJ. Cementless total hip arthroplasty performed in patients with pigmented villonodular synovitis. J Arthroplasty. 2010;25(4):552-557.
15. Chiari C, Pirich C, Brannath W, Kotz R, Trieb K. What affects the recurrence and clinical outcome of pigmented villonodular synovitis? Clin Orthop Relat Res. 2006;(450):172-178.
16. Vastel L, Lambert P, De Pinieux G, Charrois O, Kerboull M, Courpied JP. Surgical treatment of pigmented villonodular synovitis of the hip. J Bone Joint Surg Am. 2005;87(5):1019-1024.
17. Shabat S, Kollender Y, Merimsky O, et al. The use of surgery and yttrium 90 in the management of extensive and diffuse pigmented villonodular synovitis of large joints. Rheumatology. 2002;41(10):1113-1118.
18. González Della Valle A, Piccaluga F, Potter HG, Salvati EA, Pusso R. Pigmented villonodular synovitis of the hip: 2- to 23-year followup study. Clin Orthop Relat Res. 2001;(388):187-199.
19. de Visser E, Veth RP, Pruszczynski M, Wobbes T, Van de Putte LB. Diffuse and localized pigmented villonodular synovitis: evaluation of treatment of 38 patients. Arch Orthop Trauma Surg. 1999;119(7-8):401-404.
20. Aboulafia AJ, Kaplan L, Jelinek J, Benevenia J, Monson DK. Neuropathy secondary to pigmented villonodular synovitis of the hip. Clin Orthop Relat Res. 1996;(325):174-180.
21. Moroni A, Innao V, Picci P. Pigmented villonodular synovitis of the hip. Study of 9 cases. Ital J Orthop Traumatol. 1983;9(3):331-337.
22. Aglietti P, Di Muria GV, Salvati EA, Stringa G. Pigmented villonodular synovitis of the hip joint (review of the literature and report of personal case material). Ital J Orthop Traumatol. 1983;9(4):487-496.
23. Docken WP. Pigmented villonodular synovitis: a review with illustrative case reports. Semin Arthritis Rheum. 1979;9(1):1-22.
24. Scott PM. Bone lesions in pigmented villonodular synovitis. J Bone Joint Surg Br. 1968;50(2):306-311.
25. Chung SM, Janes JM. Diffuse pigmented villonodular synovitis of the hip joint. Review of the literature and report of four cases. J Bone Joint Surg Am. 1965;47:293-303.
26. McMaster PE. Pigmented villonodular synovitis with invasion of bone. Report of six cases. Rheumatology. 1960;42(7):1170-1183.
27. Ghormley RK, Romness JO. Pigmented villonodular synovitis (xanthomatosis) of the hip joint. Proc Staff Meet Mayo Clin. 1954;29(6):171-180.
28. Park KS, Diwanji SR, Yang HK, Yoon TR, Seon JK. Pigmented villonodular synovitis of the hip presenting as a buttock mass treated by total hip arthroplasty. J Arthroplasty. 2010;25(2):333.e9-e12.
29. Cowan J, Lozano-Calderón S, Ring D. Quality of prospective controlled randomized trials. Analysis of trials of treatment for lateral epicondylitis as an example. J Bone Joint Surg Am. 2007;89(8):1693-1699.
30. Harris JD, Siston RA, Pan X, Flanigan DC. Autologous chondrocyte implantation: a systematic review. J Bone Joint Surg Am. 2010;92(12):2220-2233.
31. Harris JD, Siston RA, Brophy RH, Lattermann C, Carey JL, Flanigan DC. Failures, re-operations, and complications after autologous chondrocyte implantation—a systematic review. Osteoarthritis Cartilage. 2011;19(7):779-791.
32. Rao AS, Vigorita VJ. Pigmented villonodular synovitis (giant-cell tumor of the tendon sheath and synovial membrane). A review of eighty-one cases. J Bone Joint Surg Am. 1984;66(1):76-94.
33. Kat S, Kutz R, Elbracht T, Weseloh G, Kuwert T. Radiosynovectomy in pigmented villonodular synovitis. Nuklearmedizin. 2000;39(7):209-213.
34. Gitelis S, Heligman D, Morton T. The treatment of pigmented villonodular synovitis of the hip. A case report and literature review. Clin Orthop Relat Res. 1989;(239):154-160.
35. Harris JD, McCormick FM, Abrams GD, et al. Complications and reoperations during and after hip arthroscopy: a systematic review of 92 studies and more than 6,000 patients. Arthroscopy. 2013;29(3):589-595.
Pigmented villonodular synovitis (PVNS) is a rare monoarticular disorder that affects the joints, bursae, or tendon sheaths of 1.8 per million patients.1,2 PVNS is defined by exuberant proliferation of synovial villi and nodules. Although its etiology is unknown, PVNS behaves much as a neoplastic process does, with occasional chromosomal abnormalities, local tissue invasion, and the potential for malignant transformation.3,4 Radiographs show cystic erosions or joint space narrowing, and magnetic resonance imaging shows characteristic low-signal intensity (on T1- and T2-weighted sequences) because of high hemosiderin content. Biopsy remains the gold standard for diagnosis and reveals hemosiderin-laden macrophages, vascularized villi, mononuclear cell infiltration, and sporadic mitotic figures.5 Diffuse PVNS appears as a thickened synovium with matted villi and synovial folds; localized PVNS presents as a pedunculated, firm yellow nodule.6
PVNS has a predilection for large joints, most commonly the knee (up to 80% of cases) and the hip.1,2,7 Treatment strategies for knee PVNS have been well studied and, as an aggregate, show no superiority of arthroscopic or open techniques.8 The literature on hip PVNS is less abundant and more case-based, making it difficult to reach a consensus on effective treatment. Open synovectomy and arthroplasty have been the mainstays of treatment over the past 60 years, but the advent of hip arthroscopy has introduced a new treatment modality.1,9 As arthroscopic management becomes more readily available, it is important to understand and compare the effectiveness of synovectomy and arthroplasty.
We systematically reviewed the treatment modalities for PVNS of the hip to determine how synovectomy and arthroplasty compare with respect to efficacy and revision rates.
Methods
Search Strategy
We systematically reviewed the literature according to PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) guidelines using the PRISMA checklist.10 Searches were completed in July 2014 using the PubMed Medline database and the Cochrane Central Register of Clinical Trials. Keyword selection was designed to capture all level I to V evidence English-language studies that reported clinical and/or radiographic outcomes. This was accomplished with a keyword search of all available titles and manuscript abstracts: (pigmented [Title/Abstract] AND villonodular [Title/Abstract] AND synovitis [Title/Abstract]) AND (hip [Title/Abstract]) AND (English [lang])). Abstracts from the 75 resulting studies were reviewed for exclusion criteria, which consisted of any cadaveric, biomechanical, histologic, and/or kinematic results, as well as a lack of any clinical and/or radiographic data (eg, review or technique articles). Studies were also excluded if they did not have clinical follow-up of at least 2 years. Studies not dedicated to hip PVNS specifically were not immediately excluded but were reviewed for outcomes data specific to the hip PVNS subpopulation. If a specific hip PVNS population could be distinguished from other patients, that study was included for review. If a study could not be deconstructed as such or was entirely devoted to one of our exclusion criteria, that study was excluded from our review. This initial search strategy yielded 16 studies.1,6,7,11-28
Bibliographical review of these 16 studies yielded several more for review. To ensure that no patients were counted twice, each study’s authors, data collection period, and ethnic population were reviewed and compared with those of the other studies. If there was any overlap in authorship, period, and place, only the study with the most relevant or comprehensive data was included. After accounting for all inclusion and exclusion criteria, we selected a total of 21 studies with 82 patients (86 hips) for inclusion (Figure 1).
Data Extraction
Details of study design, sample size, and patient demographics, including age, sex, and duration of symptoms, were recorded. Use of diagnostic biopsy, joint space narrowing on radiographs, treatment method, and use of radiation therapy were also abstracted. Some studies described multiple treatment methods. If those methods could not be differentiated into distinct outcomes groups, the study would have been excluded for lack of specific clinical data. Studies with sufficient data were deconstructed such that the patients from each treatment group were isolated.
Fewer than 5 studies reported physical examination findings, validated survey scores, and/or radiographic results. Therefore, the primary outcomes reported and compared between treatment groups were disease recurrence, clinical worsening defined as progressive pain or loss of function, and revision surgery. Revision surgery was subdivided into repeat synovectomy and eventual arthroplasty, arthrodesis, or revision arthroplasty. Time to revision surgery was also documented. Each study’s methodologic quality and bias were evaluated with the Modified Coleman Methodology Score (MCMS), described by Cowan and colleagues.29 MCMS is a 15-item instrument that has been used to assess randomized and nonrandomized patient trials.30,31 It has a scaled potential score ranging from 0 to 100, with scores from 85 through 100 indicating excellent, 70 through 84 good, 55 through 69 fair, and under 55 poor.
Statistical Analysis
We report our data as weighted means (SDs). A mean was calculated for each study reporting on a respective data point, and each mean was then weighted according to the sample size of that study. We multiplied each study’s individual mean by the number of patients enrolled in that study and divided the sum of all the studies’ weighted data points by the number of eligible patients in all relevant studies. The result is that the nonweighted means from studies with a smaller sample size did not carry as much weight as those from larger studies. We then compared 2 groups of patients: those who had only a synovectomy and those who had a combination of synovectomy and arthroplasty. The synovectomy-only group was also compared with a group that underwent total hip arthroplasty (THA) specifically (Figure 2). Groups were compared with Student t test (SPSS Version 18, IBM), and statistical significance was set at α = 0.05.
Results
Twenty-one studies (82 patients) were included in the final dataset (Table 1). Of these studies, 19 were retrospective case series (level IV evidence) in which the number of eligible hip PVNS patients ranged from 1 to 15. The other 2 studies were case reports (level V evidence). Mean (SD) MCMS was 25.0 (10.9).
Fifty-one patients (59.3%) were female. Mean (SD) age of all patients was 33.2 (12.6) years. Mean (SD) duration of symptoms was 4.2 (2.7) years. The right hip was affected in 59.5% of patients in whom laterality was documented. Sixty-eight patients (79.1%) had biopsy-proven PVNS; presence or absence of a biopsy was not documented for the other 18 patients.
Of the 82 patients in the study, 45 (54.9%) underwent synovectomy without arthroplasty. Staged radiation was used to augment the synovectomy in 2 of these 45 cases. One series in this group consisted of 15 cases of arthroscopic synovectomy.1 The 37 patients (45.1%) in the other treatment group had arthroplasty at time of synovectomy. These patients underwent 22 THAs, 8 cup arthroplasties, 2 metal-on-metal hip resurfacings, and 1 hemiarthroplasty. The remaining 4 patients were treated nonoperatively (3) or with primary arthrodesis (1).
Comparisons between the synovectomy-only and synovectomy-with-arthroplasty groups are listed in Table 2. Synovectomy patients were younger on average than arthroplasty patients, but the difference was not statistically significant (P = .28). Only 6 studies distinguished between local and diffuse PVNS histology, and the diffuse type was detected in 87.0%, with insufficient data to detect a difference between the synovectomy and arthroplasty groups. In studies with documented radiographic findings, 75.0% of patients had evidence of joint space narrowing, which was significantly (P = .03) more common in the arthroplasty group (96.7% vs 31.3%).
Mean (SD) clinical follow-up was 8.4 (5.9) years for all patients. A larger percentage of synovectomy-only patients experienced recurrence and worsened symptoms, but neither trend achieved statistical significance. The rate of eventual THA or arthrodesis after synovectomy alone was almost identical (P = .17) to the rate of revision THA in the synovectomy-with-arthroplasty group (26.2% vs 24.3%). Time to revision surgery, however, was significantly (P = .02) longer in the arthroplasty group. Two additional patients in the synovectomy-with-arthroplasty group underwent repeat synovectomy alone, but no patients in the synovectomy-only group underwent repeat synovectomy without arthroplasty.
One nonoperatively managed patient experienced symptom progression over the course of 10 years. The other 2 patients were stable after 2- and 4-year follow-up. The arthrodesis patient did not experience recurrence or have a revision operation in the 5 years after the index procedure.
Discussion
PVNS is a proliferative disorder of synovial tissue with a high risk of recurrence.15,32 Metastasis is extremely rare; there is only 1 case report of a fatality, which occurred within 42 months.12 Chiari and colleagues15 suggested that the PVNS recurrence rate is highest in the large joints. Therefore, in hip PVNS, early surgical resection is needed to limit articular destruction and the potential for recurrence. The primary treatment modalities are synovectomy alone and synovectomy with arthroplasty, which includes THA, cup arthroplasty, hip resurfacing, and hemiarthroplasty. According to our systematic review, about one-fourth of all patients in both treatment groups ultimately underwent revision surgery. Mean time to revision was significantly longer for synovectomy-with-arthroplasty patients (almost 12 years) than for synovectomy-only patients (6.5 years). One potential explanation is that arthroplasty component fixation may take longer to loosen than an inadequately synovectomized joint takes to recur. The synovectomy-only group did have a higher recurrence rate, though the difference was not statistically significant.
Open synovectomy is the most widely described technique for addressing hip PVNS. The precise pathophysiology of PVNS remains largely unknown, but most authors agree that aggressive débridement is required to halt its locally invasive course. Scott24 described the invasion of vascular foramina from synovium into bone and thought that radical synovectomy was essential to remove the stalks of these synovial villi. Furthermore, PVNS most commonly affects adults in the third through fifth decades of life,7 and many surgeons want to avoid prosthetic components (which may loosen over time) in this age group. Synovectomy, however, has persistently high recurrence rates, and, without removal of the femoral head and neck, it can be difficult to obtain adequate exposure for complete débridement. Although adjuvant external beam radiation has been used by some authors,17,19,33 its utility is unproven, and other authors have cautioned against unnecessary irradiation of reproductive organs.1,24,34
The high rates of bony involvement, joint destruction, and recurrence after synovectomy have prompted many surgeons to turn to arthroplasty. González Della Valle and colleagues18 theorized that joint space narrowing is more common in hip PVNS because of the poor distensibility of the hip capsule compared with that of the knee and other joints. In turn, bony lesions and arthritis present earlier in hip PVNS.14 Yoo and colleagues14 found a statistically significant increase in Harris Hip Scale (HHS) scores and a high rate of return to athletic activity after THA for PVNS. However, they also reported revisions for component loosening and osteolysis in 2 of 8 patients and periprosthetic osteolysis without loosening in another 2 patients. Vastel and colleagues16 similarly reported aseptic loosening of the acetabular component in half their patient cohort. No studies have determined which condition—PVNS recurrence or debris-related osteolysis—causes the accelerated loosening in this demographic.
Byrd and colleagues1 recently described use of hip arthroscopy in the treatment of PVNS. In a cohort of 13 patients, they found statistically significant improvements in HHS scores, no postoperative complications, and only 1 revision (THA 6 years after surgery). Although there is a prevailing perception that nodular (vs diffuse) PVNS is more appropriately treated with arthroscopic excision, no studies have provided data on this effect, and Byrd and colleagues1 in fact showed a trend of slightly better outcomes in diffuse cases than in nodular cases. The main challenges of hip arthroscopy are the steep learning curve and adequate exposure. Recent innovations include additional arthroscopic portals and enlarged T-capsulotomy, which may be contributing to decreased complication rates in hip arthroscopy in general.35
The limitations of this systematic review were largely imposed by the studies analyzed. The primary limitation was the relative paucity of clinical and radiographic data on hip PVNS. To our knowledge, studies on the treatment of hip PVNS have reported evidence levels no higher than IV. In addition, the studies we reviewed often had only 1 or 2 patient cases satisfying our inclusion criteria. For this reason, we included case reports, which further lowered the level of evidence of studies used. There were no consistently reported physical examination, survey, or radiographic findings that could be used to compare studies. All studies with sufficient data on hip PVNS treatment outcomes were rated poorly with the Modified Coleman Methodology Scoring system.29 Selection bias was minimized by the inclusive nature of studies with level I to V evidence, but this led to a study design bias in that most studies consisted of level IV evidence.
Conclusion
Although the hip PVNS literature is limited, our review provides insight into expected outcomes. No matter which surgery is to be performed, surgeons must counsel patients about the high revision rate. One in 4 patients ultimately undergoes a second surgery, which may be required within 6 or 7 years after synovectomy without arthroplasty. Further development and innovation in hip arthroscopy may transform the treatment of PVNS. We encourage other investigators to conduct prospective, comparative trials with higher evidence levels to assess the utility of arthroscopy and other treatment modalities.
Pigmented villonodular synovitis (PVNS) is a rare monoarticular disorder that affects the joints, bursae, or tendon sheaths of 1.8 per million patients.1,2 PVNS is defined by exuberant proliferation of synovial villi and nodules. Although its etiology is unknown, PVNS behaves much as a neoplastic process does, with occasional chromosomal abnormalities, local tissue invasion, and the potential for malignant transformation.3,4 Radiographs show cystic erosions or joint space narrowing, and magnetic resonance imaging shows characteristic low-signal intensity (on T1- and T2-weighted sequences) because of high hemosiderin content. Biopsy remains the gold standard for diagnosis and reveals hemosiderin-laden macrophages, vascularized villi, mononuclear cell infiltration, and sporadic mitotic figures.5 Diffuse PVNS appears as a thickened synovium with matted villi and synovial folds; localized PVNS presents as a pedunculated, firm yellow nodule.6
PVNS has a predilection for large joints, most commonly the knee (up to 80% of cases) and the hip.1,2,7 Treatment strategies for knee PVNS have been well studied and, as an aggregate, show no superiority of arthroscopic or open techniques.8 The literature on hip PVNS is less abundant and more case-based, making it difficult to reach a consensus on effective treatment. Open synovectomy and arthroplasty have been the mainstays of treatment over the past 60 years, but the advent of hip arthroscopy has introduced a new treatment modality.1,9 As arthroscopic management becomes more readily available, it is important to understand and compare the effectiveness of synovectomy and arthroplasty.
We systematically reviewed the treatment modalities for PVNS of the hip to determine how synovectomy and arthroplasty compare with respect to efficacy and revision rates.
Methods
Search Strategy
We systematically reviewed the literature according to PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) guidelines using the PRISMA checklist.10 Searches were completed in July 2014 using the PubMed Medline database and the Cochrane Central Register of Clinical Trials. Keyword selection was designed to capture all level I to V evidence English-language studies that reported clinical and/or radiographic outcomes. This was accomplished with a keyword search of all available titles and manuscript abstracts: (pigmented [Title/Abstract] AND villonodular [Title/Abstract] AND synovitis [Title/Abstract]) AND (hip [Title/Abstract]) AND (English [lang])). Abstracts from the 75 resulting studies were reviewed for exclusion criteria, which consisted of any cadaveric, biomechanical, histologic, and/or kinematic results, as well as a lack of any clinical and/or radiographic data (eg, review or technique articles). Studies were also excluded if they did not have clinical follow-up of at least 2 years. Studies not dedicated to hip PVNS specifically were not immediately excluded but were reviewed for outcomes data specific to the hip PVNS subpopulation. If a specific hip PVNS population could be distinguished from other patients, that study was included for review. If a study could not be deconstructed as such or was entirely devoted to one of our exclusion criteria, that study was excluded from our review. This initial search strategy yielded 16 studies.1,6,7,11-28
Bibliographical review of these 16 studies yielded several more for review. To ensure that no patients were counted twice, each study’s authors, data collection period, and ethnic population were reviewed and compared with those of the other studies. If there was any overlap in authorship, period, and place, only the study with the most relevant or comprehensive data was included. After accounting for all inclusion and exclusion criteria, we selected a total of 21 studies with 82 patients (86 hips) for inclusion (Figure 1).
Data Extraction
Details of study design, sample size, and patient demographics, including age, sex, and duration of symptoms, were recorded. Use of diagnostic biopsy, joint space narrowing on radiographs, treatment method, and use of radiation therapy were also abstracted. Some studies described multiple treatment methods. If those methods could not be differentiated into distinct outcomes groups, the study would have been excluded for lack of specific clinical data. Studies with sufficient data were deconstructed such that the patients from each treatment group were isolated.
Fewer than 5 studies reported physical examination findings, validated survey scores, and/or radiographic results. Therefore, the primary outcomes reported and compared between treatment groups were disease recurrence, clinical worsening defined as progressive pain or loss of function, and revision surgery. Revision surgery was subdivided into repeat synovectomy and eventual arthroplasty, arthrodesis, or revision arthroplasty. Time to revision surgery was also documented. Each study’s methodologic quality and bias were evaluated with the Modified Coleman Methodology Score (MCMS), described by Cowan and colleagues.29 MCMS is a 15-item instrument that has been used to assess randomized and nonrandomized patient trials.30,31 It has a scaled potential score ranging from 0 to 100, with scores from 85 through 100 indicating excellent, 70 through 84 good, 55 through 69 fair, and under 55 poor.
Statistical Analysis
We report our data as weighted means (SDs). A mean was calculated for each study reporting on a respective data point, and each mean was then weighted according to the sample size of that study. We multiplied each study’s individual mean by the number of patients enrolled in that study and divided the sum of all the studies’ weighted data points by the number of eligible patients in all relevant studies. The result is that the nonweighted means from studies with a smaller sample size did not carry as much weight as those from larger studies. We then compared 2 groups of patients: those who had only a synovectomy and those who had a combination of synovectomy and arthroplasty. The synovectomy-only group was also compared with a group that underwent total hip arthroplasty (THA) specifically (Figure 2). Groups were compared with Student t test (SPSS Version 18, IBM), and statistical significance was set at α = 0.05.
Results
Twenty-one studies (82 patients) were included in the final dataset (Table 1). Of these studies, 19 were retrospective case series (level IV evidence) in which the number of eligible hip PVNS patients ranged from 1 to 15. The other 2 studies were case reports (level V evidence). Mean (SD) MCMS was 25.0 (10.9).
Fifty-one patients (59.3%) were female. Mean (SD) age of all patients was 33.2 (12.6) years. Mean (SD) duration of symptoms was 4.2 (2.7) years. The right hip was affected in 59.5% of patients in whom laterality was documented. Sixty-eight patients (79.1%) had biopsy-proven PVNS; presence or absence of a biopsy was not documented for the other 18 patients.
Of the 82 patients in the study, 45 (54.9%) underwent synovectomy without arthroplasty. Staged radiation was used to augment the synovectomy in 2 of these 45 cases. One series in this group consisted of 15 cases of arthroscopic synovectomy.1 The 37 patients (45.1%) in the other treatment group had arthroplasty at time of synovectomy. These patients underwent 22 THAs, 8 cup arthroplasties, 2 metal-on-metal hip resurfacings, and 1 hemiarthroplasty. The remaining 4 patients were treated nonoperatively (3) or with primary arthrodesis (1).
Comparisons between the synovectomy-only and synovectomy-with-arthroplasty groups are listed in Table 2. Synovectomy patients were younger on average than arthroplasty patients, but the difference was not statistically significant (P = .28). Only 6 studies distinguished between local and diffuse PVNS histology, and the diffuse type was detected in 87.0%, with insufficient data to detect a difference between the synovectomy and arthroplasty groups. In studies with documented radiographic findings, 75.0% of patients had evidence of joint space narrowing, which was significantly (P = .03) more common in the arthroplasty group (96.7% vs 31.3%).
Mean (SD) clinical follow-up was 8.4 (5.9) years for all patients. A larger percentage of synovectomy-only patients experienced recurrence and worsened symptoms, but neither trend achieved statistical significance. The rate of eventual THA or arthrodesis after synovectomy alone was almost identical (P = .17) to the rate of revision THA in the synovectomy-with-arthroplasty group (26.2% vs 24.3%). Time to revision surgery, however, was significantly (P = .02) longer in the arthroplasty group. Two additional patients in the synovectomy-with-arthroplasty group underwent repeat synovectomy alone, but no patients in the synovectomy-only group underwent repeat synovectomy without arthroplasty.
One nonoperatively managed patient experienced symptom progression over the course of 10 years. The other 2 patients were stable after 2- and 4-year follow-up. The arthrodesis patient did not experience recurrence or have a revision operation in the 5 years after the index procedure.
Discussion
PVNS is a proliferative disorder of synovial tissue with a high risk of recurrence.15,32 Metastasis is extremely rare; there is only 1 case report of a fatality, which occurred within 42 months.12 Chiari and colleagues15 suggested that the PVNS recurrence rate is highest in the large joints. Therefore, in hip PVNS, early surgical resection is needed to limit articular destruction and the potential for recurrence. The primary treatment modalities are synovectomy alone and synovectomy with arthroplasty, which includes THA, cup arthroplasty, hip resurfacing, and hemiarthroplasty. According to our systematic review, about one-fourth of all patients in both treatment groups ultimately underwent revision surgery. Mean time to revision was significantly longer for synovectomy-with-arthroplasty patients (almost 12 years) than for synovectomy-only patients (6.5 years). One potential explanation is that arthroplasty component fixation may take longer to loosen than an inadequately synovectomized joint takes to recur. The synovectomy-only group did have a higher recurrence rate, though the difference was not statistically significant.
Open synovectomy is the most widely described technique for addressing hip PVNS. The precise pathophysiology of PVNS remains largely unknown, but most authors agree that aggressive débridement is required to halt its locally invasive course. Scott24 described the invasion of vascular foramina from synovium into bone and thought that radical synovectomy was essential to remove the stalks of these synovial villi. Furthermore, PVNS most commonly affects adults in the third through fifth decades of life,7 and many surgeons want to avoid prosthetic components (which may loosen over time) in this age group. Synovectomy, however, has persistently high recurrence rates, and, without removal of the femoral head and neck, it can be difficult to obtain adequate exposure for complete débridement. Although adjuvant external beam radiation has been used by some authors,17,19,33 its utility is unproven, and other authors have cautioned against unnecessary irradiation of reproductive organs.1,24,34
The high rates of bony involvement, joint destruction, and recurrence after synovectomy have prompted many surgeons to turn to arthroplasty. González Della Valle and colleagues18 theorized that joint space narrowing is more common in hip PVNS because of the poor distensibility of the hip capsule compared with that of the knee and other joints. In turn, bony lesions and arthritis present earlier in hip PVNS.14 Yoo and colleagues14 found a statistically significant increase in Harris Hip Scale (HHS) scores and a high rate of return to athletic activity after THA for PVNS. However, they also reported revisions for component loosening and osteolysis in 2 of 8 patients and periprosthetic osteolysis without loosening in another 2 patients. Vastel and colleagues16 similarly reported aseptic loosening of the acetabular component in half their patient cohort. No studies have determined which condition—PVNS recurrence or debris-related osteolysis—causes the accelerated loosening in this demographic.
Byrd and colleagues1 recently described use of hip arthroscopy in the treatment of PVNS. In a cohort of 13 patients, they found statistically significant improvements in HHS scores, no postoperative complications, and only 1 revision (THA 6 years after surgery). Although there is a prevailing perception that nodular (vs diffuse) PVNS is more appropriately treated with arthroscopic excision, no studies have provided data on this effect, and Byrd and colleagues1 in fact showed a trend of slightly better outcomes in diffuse cases than in nodular cases. The main challenges of hip arthroscopy are the steep learning curve and adequate exposure. Recent innovations include additional arthroscopic portals and enlarged T-capsulotomy, which may be contributing to decreased complication rates in hip arthroscopy in general.35
The limitations of this systematic review were largely imposed by the studies analyzed. The primary limitation was the relative paucity of clinical and radiographic data on hip PVNS. To our knowledge, studies on the treatment of hip PVNS have reported evidence levels no higher than IV. In addition, the studies we reviewed often had only 1 or 2 patient cases satisfying our inclusion criteria. For this reason, we included case reports, which further lowered the level of evidence of studies used. There were no consistently reported physical examination, survey, or radiographic findings that could be used to compare studies. All studies with sufficient data on hip PVNS treatment outcomes were rated poorly with the Modified Coleman Methodology Scoring system.29 Selection bias was minimized by the inclusive nature of studies with level I to V evidence, but this led to a study design bias in that most studies consisted of level IV evidence.
Conclusion
Although the hip PVNS literature is limited, our review provides insight into expected outcomes. No matter which surgery is to be performed, surgeons must counsel patients about the high revision rate. One in 4 patients ultimately undergoes a second surgery, which may be required within 6 or 7 years after synovectomy without arthroplasty. Further development and innovation in hip arthroscopy may transform the treatment of PVNS. We encourage other investigators to conduct prospective, comparative trials with higher evidence levels to assess the utility of arthroscopy and other treatment modalities.
1. Byrd JWT, Jones KS, Maiers GP. Two to 10 years’ follow-up of arthroscopic management of pigmented villonodular synovitis in the hip: a case series. Arthroscopy. 2013;29(11):1783-1787.
2. Myers BW, Masi AT. Pigmented villonodular synovitis and tenosynovitis: a clinical epidemiologic study of 166 cases and literature review. Medicine. 1980;59(3):223-238.
3. Sciot R, Rosai J, Dal Cin P, et al. Analysis of 35 cases of localized and diffuse tenosynovial giant cell tumor: a report from the Chromosomes and Morphology (CHAMP) study group. Mod Pathol. 1999;12(6):576-579.
4. Bertoni F, Unni KK, Beabout JW, Sim FH. Malignant giant cell tumor of the tendon sheaths and joints (malignant pigmented villonodular synovitis). Am J Surg Pathol. 1997;21(2):153-163.
5. Mankin H, Trahan C, Hornicek F. Pigmented villonodular synovitis of joints. J Surg Oncol. 2011;103(5):386-389.
6. Martin RC, Osborne DL, Edwards MJ, Wrightson W, McMasters KM. Giant cell tumor of tendon sheath, tenosynovial giant cell tumor, and pigmented villonodular synovitis: defining the presentation, surgical therapy and recurrence. Oncol Rep. 2000;7(2):413-419.
7. Danzig LA, Gershuni DH, Resnick D. Diagnosis and treatment of diffuse pigmented villonodular synovitis of the hip. Clin Orthop Relat Res. 1982;(168):42-47.
8. Aurégan JC, Klouche S, Bohu Y, Lefèvre N, Herman S, Hardy P. Treatment of pigmented villonodular synovitis of the knee. Arthroscopy. 2014;30(10):1327-1341.
9. Gondolph-Zink B, Puhl W, Noack W. Semiarthroscopic synovectomy of the hip. Int Orthop. 1988;12(1):31-35.
10. Moher D, Liberati A, Tetzlaff J, Altman DG; PRISMA Group. Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. J Clin Epidemiol. 2009;62(10):1006-1012.
11. Shoji T, Yasunaga Y, Yamasaki T, et al. Transtrochanteric rotational osteotomy combined with intra-articular procedures for pigmented villonodular synovitis of the hip. J Orthop Sci. 2015;20(5):943-950.
12. Li LM, Jeffery J. Exceptionally aggressive pigmented villonodular synovitis of the hip unresponsive to radiotherapy. J Bone Joint Surg Br. 2011;93(7):995-997.
13. Hoberg M, Amstutz HC. Metal-on-metal hip resurfacing in patients with pigmented villonodular synovitis: a report of two cases. Orthopedics. 2010;33(1):50-53.
14. Yoo JJ, Kwon YS, Koo KH, Yoon KS, Min BW, Kim HJ. Cementless total hip arthroplasty performed in patients with pigmented villonodular synovitis. J Arthroplasty. 2010;25(4):552-557.
15. Chiari C, Pirich C, Brannath W, Kotz R, Trieb K. What affects the recurrence and clinical outcome of pigmented villonodular synovitis? Clin Orthop Relat Res. 2006;(450):172-178.
16. Vastel L, Lambert P, De Pinieux G, Charrois O, Kerboull M, Courpied JP. Surgical treatment of pigmented villonodular synovitis of the hip. J Bone Joint Surg Am. 2005;87(5):1019-1024.
17. Shabat S, Kollender Y, Merimsky O, et al. The use of surgery and yttrium 90 in the management of extensive and diffuse pigmented villonodular synovitis of large joints. Rheumatology. 2002;41(10):1113-1118.
18. González Della Valle A, Piccaluga F, Potter HG, Salvati EA, Pusso R. Pigmented villonodular synovitis of the hip: 2- to 23-year followup study. Clin Orthop Relat Res. 2001;(388):187-199.
19. de Visser E, Veth RP, Pruszczynski M, Wobbes T, Van de Putte LB. Diffuse and localized pigmented villonodular synovitis: evaluation of treatment of 38 patients. Arch Orthop Trauma Surg. 1999;119(7-8):401-404.
20. Aboulafia AJ, Kaplan L, Jelinek J, Benevenia J, Monson DK. Neuropathy secondary to pigmented villonodular synovitis of the hip. Clin Orthop Relat Res. 1996;(325):174-180.
21. Moroni A, Innao V, Picci P. Pigmented villonodular synovitis of the hip. Study of 9 cases. Ital J Orthop Traumatol. 1983;9(3):331-337.
22. Aglietti P, Di Muria GV, Salvati EA, Stringa G. Pigmented villonodular synovitis of the hip joint (review of the literature and report of personal case material). Ital J Orthop Traumatol. 1983;9(4):487-496.
23. Docken WP. Pigmented villonodular synovitis: a review with illustrative case reports. Semin Arthritis Rheum. 1979;9(1):1-22.
24. Scott PM. Bone lesions in pigmented villonodular synovitis. J Bone Joint Surg Br. 1968;50(2):306-311.
25. Chung SM, Janes JM. Diffuse pigmented villonodular synovitis of the hip joint. Review of the literature and report of four cases. J Bone Joint Surg Am. 1965;47:293-303.
26. McMaster PE. Pigmented villonodular synovitis with invasion of bone. Report of six cases. Rheumatology. 1960;42(7):1170-1183.
27. Ghormley RK, Romness JO. Pigmented villonodular synovitis (xanthomatosis) of the hip joint. Proc Staff Meet Mayo Clin. 1954;29(6):171-180.
28. Park KS, Diwanji SR, Yang HK, Yoon TR, Seon JK. Pigmented villonodular synovitis of the hip presenting as a buttock mass treated by total hip arthroplasty. J Arthroplasty. 2010;25(2):333.e9-e12.
29. Cowan J, Lozano-Calderón S, Ring D. Quality of prospective controlled randomized trials. Analysis of trials of treatment for lateral epicondylitis as an example. J Bone Joint Surg Am. 2007;89(8):1693-1699.
30. Harris JD, Siston RA, Pan X, Flanigan DC. Autologous chondrocyte implantation: a systematic review. J Bone Joint Surg Am. 2010;92(12):2220-2233.
31. Harris JD, Siston RA, Brophy RH, Lattermann C, Carey JL, Flanigan DC. Failures, re-operations, and complications after autologous chondrocyte implantation—a systematic review. Osteoarthritis Cartilage. 2011;19(7):779-791.
32. Rao AS, Vigorita VJ. Pigmented villonodular synovitis (giant-cell tumor of the tendon sheath and synovial membrane). A review of eighty-one cases. J Bone Joint Surg Am. 1984;66(1):76-94.
33. Kat S, Kutz R, Elbracht T, Weseloh G, Kuwert T. Radiosynovectomy in pigmented villonodular synovitis. Nuklearmedizin. 2000;39(7):209-213.
34. Gitelis S, Heligman D, Morton T. The treatment of pigmented villonodular synovitis of the hip. A case report and literature review. Clin Orthop Relat Res. 1989;(239):154-160.
35. Harris JD, McCormick FM, Abrams GD, et al. Complications and reoperations during and after hip arthroscopy: a systematic review of 92 studies and more than 6,000 patients. Arthroscopy. 2013;29(3):589-595.
1. Byrd JWT, Jones KS, Maiers GP. Two to 10 years’ follow-up of arthroscopic management of pigmented villonodular synovitis in the hip: a case series. Arthroscopy. 2013;29(11):1783-1787.
2. Myers BW, Masi AT. Pigmented villonodular synovitis and tenosynovitis: a clinical epidemiologic study of 166 cases and literature review. Medicine. 1980;59(3):223-238.
3. Sciot R, Rosai J, Dal Cin P, et al. Analysis of 35 cases of localized and diffuse tenosynovial giant cell tumor: a report from the Chromosomes and Morphology (CHAMP) study group. Mod Pathol. 1999;12(6):576-579.
4. Bertoni F, Unni KK, Beabout JW, Sim FH. Malignant giant cell tumor of the tendon sheaths and joints (malignant pigmented villonodular synovitis). Am J Surg Pathol. 1997;21(2):153-163.
5. Mankin H, Trahan C, Hornicek F. Pigmented villonodular synovitis of joints. J Surg Oncol. 2011;103(5):386-389.
6. Martin RC, Osborne DL, Edwards MJ, Wrightson W, McMasters KM. Giant cell tumor of tendon sheath, tenosynovial giant cell tumor, and pigmented villonodular synovitis: defining the presentation, surgical therapy and recurrence. Oncol Rep. 2000;7(2):413-419.
7. Danzig LA, Gershuni DH, Resnick D. Diagnosis and treatment of diffuse pigmented villonodular synovitis of the hip. Clin Orthop Relat Res. 1982;(168):42-47.
8. Aurégan JC, Klouche S, Bohu Y, Lefèvre N, Herman S, Hardy P. Treatment of pigmented villonodular synovitis of the knee. Arthroscopy. 2014;30(10):1327-1341.
9. Gondolph-Zink B, Puhl W, Noack W. Semiarthroscopic synovectomy of the hip. Int Orthop. 1988;12(1):31-35.
10. Moher D, Liberati A, Tetzlaff J, Altman DG; PRISMA Group. Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. J Clin Epidemiol. 2009;62(10):1006-1012.
11. Shoji T, Yasunaga Y, Yamasaki T, et al. Transtrochanteric rotational osteotomy combined with intra-articular procedures for pigmented villonodular synovitis of the hip. J Orthop Sci. 2015;20(5):943-950.
12. Li LM, Jeffery J. Exceptionally aggressive pigmented villonodular synovitis of the hip unresponsive to radiotherapy. J Bone Joint Surg Br. 2011;93(7):995-997.
13. Hoberg M, Amstutz HC. Metal-on-metal hip resurfacing in patients with pigmented villonodular synovitis: a report of two cases. Orthopedics. 2010;33(1):50-53.
14. Yoo JJ, Kwon YS, Koo KH, Yoon KS, Min BW, Kim HJ. Cementless total hip arthroplasty performed in patients with pigmented villonodular synovitis. J Arthroplasty. 2010;25(4):552-557.
15. Chiari C, Pirich C, Brannath W, Kotz R, Trieb K. What affects the recurrence and clinical outcome of pigmented villonodular synovitis? Clin Orthop Relat Res. 2006;(450):172-178.
16. Vastel L, Lambert P, De Pinieux G, Charrois O, Kerboull M, Courpied JP. Surgical treatment of pigmented villonodular synovitis of the hip. J Bone Joint Surg Am. 2005;87(5):1019-1024.
17. Shabat S, Kollender Y, Merimsky O, et al. The use of surgery and yttrium 90 in the management of extensive and diffuse pigmented villonodular synovitis of large joints. Rheumatology. 2002;41(10):1113-1118.
18. González Della Valle A, Piccaluga F, Potter HG, Salvati EA, Pusso R. Pigmented villonodular synovitis of the hip: 2- to 23-year followup study. Clin Orthop Relat Res. 2001;(388):187-199.
19. de Visser E, Veth RP, Pruszczynski M, Wobbes T, Van de Putte LB. Diffuse and localized pigmented villonodular synovitis: evaluation of treatment of 38 patients. Arch Orthop Trauma Surg. 1999;119(7-8):401-404.
20. Aboulafia AJ, Kaplan L, Jelinek J, Benevenia J, Monson DK. Neuropathy secondary to pigmented villonodular synovitis of the hip. Clin Orthop Relat Res. 1996;(325):174-180.
21. Moroni A, Innao V, Picci P. Pigmented villonodular synovitis of the hip. Study of 9 cases. Ital J Orthop Traumatol. 1983;9(3):331-337.
22. Aglietti P, Di Muria GV, Salvati EA, Stringa G. Pigmented villonodular synovitis of the hip joint (review of the literature and report of personal case material). Ital J Orthop Traumatol. 1983;9(4):487-496.
23. Docken WP. Pigmented villonodular synovitis: a review with illustrative case reports. Semin Arthritis Rheum. 1979;9(1):1-22.
24. Scott PM. Bone lesions in pigmented villonodular synovitis. J Bone Joint Surg Br. 1968;50(2):306-311.
25. Chung SM, Janes JM. Diffuse pigmented villonodular synovitis of the hip joint. Review of the literature and report of four cases. J Bone Joint Surg Am. 1965;47:293-303.
26. McMaster PE. Pigmented villonodular synovitis with invasion of bone. Report of six cases. Rheumatology. 1960;42(7):1170-1183.
27. Ghormley RK, Romness JO. Pigmented villonodular synovitis (xanthomatosis) of the hip joint. Proc Staff Meet Mayo Clin. 1954;29(6):171-180.
28. Park KS, Diwanji SR, Yang HK, Yoon TR, Seon JK. Pigmented villonodular synovitis of the hip presenting as a buttock mass treated by total hip arthroplasty. J Arthroplasty. 2010;25(2):333.e9-e12.
29. Cowan J, Lozano-Calderón S, Ring D. Quality of prospective controlled randomized trials. Analysis of trials of treatment for lateral epicondylitis as an example. J Bone Joint Surg Am. 2007;89(8):1693-1699.
30. Harris JD, Siston RA, Pan X, Flanigan DC. Autologous chondrocyte implantation: a systematic review. J Bone Joint Surg Am. 2010;92(12):2220-2233.
31. Harris JD, Siston RA, Brophy RH, Lattermann C, Carey JL, Flanigan DC. Failures, re-operations, and complications after autologous chondrocyte implantation—a systematic review. Osteoarthritis Cartilage. 2011;19(7):779-791.
32. Rao AS, Vigorita VJ. Pigmented villonodular synovitis (giant-cell tumor of the tendon sheath and synovial membrane). A review of eighty-one cases. J Bone Joint Surg Am. 1984;66(1):76-94.
33. Kat S, Kutz R, Elbracht T, Weseloh G, Kuwert T. Radiosynovectomy in pigmented villonodular synovitis. Nuklearmedizin. 2000;39(7):209-213.
34. Gitelis S, Heligman D, Morton T. The treatment of pigmented villonodular synovitis of the hip. A case report and literature review. Clin Orthop Relat Res. 1989;(239):154-160.
35. Harris JD, McCormick FM, Abrams GD, et al. Complications and reoperations during and after hip arthroscopy: a systematic review of 92 studies and more than 6,000 patients. Arthroscopy. 2013;29(3):589-595.
Debunking marijuana myths for teens
The annual checkup has long provided an opportunity for early adolescents to learn about the risks of alcohol and drug use from a trusted source who may be less biased than parents, teachers, or police. Parents also turn to their child’s pediatrician for guidance on how to broach this important topic with their children, or they may come with concerns about their children’s use of drugs or alcohol.
Marijuana has become an increasingly complex topic, as its legal status has rapidly changed: It’s legal to purchase marijuana in four states (Alaska, Colorado, Oregon, and Washington, as well as the District of Columbia); it is decriminalized in 20 states and the District of Columbia for certain marijuana possession offenses; and it is legal to use medical marijuana in 23 states. As its legal status changes, attitudes about its use also have shifted, and its availability, form, and potency all have changed dramatically in just the past decade. Further, we ourselves may have mixed feelings about marijuana use based on our own experience as adolescents and sampling bias. We may have seen its low-level use and minimal effects in young or mature adults, or we may have seen substantial use of marijuana have a major deleterious impact on a friend or become a gateway drug for addiction to dangerous substances.
Before addressing marijuana use with adolescent patients and dealing with their potential skepticism concerning any harm, it is worth spending a little time looking in the mirror to consider your perspective on marijuana use and your response to disbelief.
According to the National Institute on Drug Abuse’s Monitoring the Future (MTF) survey, almost 12% of 8th graders, 27% of 10th graders, and 35% of 12th graders in the United States reported having used marijuana in the past year. Among the 12th graders in that 2014 survey, almost 20% were current users of marijuana and 6% were daily users. Many surveys, including the MTF, have demonstrated that attitudes of teenagers have shifted about marijuana’s dangerousness, with a steep and steady decline in the number of teenagers believing that regular marijuana use poses a risk to their health and well-being. In 2014, less than 40% of 12th graders in the MTF survey agreed that regular use of marijuana would pose a risk to their well-being, compared with a peak of almost 80% of 12th graders in the early 1990’s.
Pediatricians have an opportunity to change their patient’s thinking about marijuana. At the checkup when you routinely ask about alcohol and drug use, ask about marijuana use in particular. You might start by asking if they have heard their friends talking about marijuana? What have they heard? Are other kids using it? Have they ever seen anyone use it? Have their friends invited them to try? You should find out if they think it is safe or dangerous, and how it compares with cigarettes, alcohol, and other drugs on this score. Then you may be able to debunk some myths you hear from them.
Myth No. 1: Marijuana is medicine
Although 23 states allow the legal sale of marijuana for “medicinal purposes,” it is important to note that there are currently no Food and Drug Administration–approved indications for medical marijuana. There is modest evidence that the active compounds in marijuana (delta-9-tetra-hydrocannabinol [THC] and other cannabinoids) can be effective in the management of the muscle spasticity associated with multiple sclerosis, the treatment of nausea associated with chemotherapy, and increasing the appetite of patients with wasting due to AIDS, and there are FDA-approved synthetic cannabinoids that can be prescribed for these symptoms. It is also important to note that there is no evidence that THC or other cannabinoids are useful in the treatment of mood or anxiety symptoms, even though these are often used as reasons for seeking medicinal marijuana. Indeed, marijuana may cause or worsen several psychiatric problems.
Myth No. 2: Marijuana is safe
Although there is consensus that moderate marijuana use in adulthood poses only limited health risks (including the known risks of smoking), there is robust evidence that marijuana use during youth (through the early 20s) causes several serious and permanent effects on the developing brain. One 2012 study showed that for youth who are dependent on marijuana before they are 18 years, there is an 8-point drop in IQ in adulthood (Proc Natl Acad Sci USA. 2012 Oct 2;109[40]:E2657-64). This IQ drop persists even if they quit smoking, and does not occur for those who first become dependent on marijuana in adulthood. A 2015 study demonstrated that even for adolescents who are light smokers (one to two times weekly) with no evidence of marijuana dependence, there are significant abnormalities in the size and shape of their amygdala and nucleus accumbens, with associated changes in their motivation, decision making, attention, functional memory, and processing of emotions(J Neurosci. 2014 Apr 16;34[16]:5529-38). These abnormalities increase with increased frequency of use, and are not seen in those who begin smoking in adulthood (mid-20s and later).
Beyond these findings of cognitive deficits, evidence is growing that adolescent marijuana use is associated with several psychiatric illnesses, including depression and anxiety. There is especially strong evidence for a causal link between marijuana use and psychotic illnesses in (genetically) vulnerable young people. Any marijuana user can experience a brief psychotic reaction if the amount ingested or smoked is great enough, but for those young people who carry a specific variant of the gene for catechol-o-methyltransferase (COMT, an enzyme that degrades neurotransmitters), smoking marijuana in adolescence nearly triples their risk of developing schizophrenia in adulthood. For youth with a variant of the AKT gene (another enzyme affecting dopamine signaling in the brain), daily use of marijuana raises their risk of developing schizophrenia sevenfold. Clearly, marijuana can be the critical environmental trigger for schizophrenia in genetically vulnerable youth. Until we have a comprehensive knowledge of the relevant genes, and routinely check every patient’s complete genetic profile, it is reasonable to assume that any young person using marijuana is significantly increasing the risk of developing schizophrenia, a chronic and disabling condition.
Myth No. 3: Marijuana has no effect on driving
Marijuana intoxication significantly affects motor coordination, reaction time, and judgment, and multiple studies have demonstrated a direct relationship between blood THC concentration and impaired driving ability. A recent meta-analysis demonstrated that the risk of being in a car accident doubled after marijuana use (Drug Alcohol Depend. 2004 Feb 7;73[2]:109-19). These studies usually involved adults, and it is reasonable to assume that the risks may be more pronounced in adolescents, particularly ones who are new to driving or have other problems that could affect their attention or reaction time (such as attention-deficit/hyperactivity disorder). Beyond letting patients know about the increased risks of accidents, it may be worth reminding them that driving while intoxicated – even with legal use marijuana – is a criminal offense.
Myth No. 4: Marijuana has no effect on schoolwork
Aside from the risks of causing long-term cognitive changes and psychiatric problems that can affect school performance, the direct effects of marijuana intoxication can linger and affect school performance well after its use. The “high” from marijuana typically lasts from 1 to 3 hours, but the drug’s effects on higher-level cognitive processes (mediated by the neocortex and hippocampus) can last for days. So a teenager who smokes on Saturday night may have lingering impairment of motivation, the ability to shift attention, the ability to learn complex tasks, and working memory. These are all critical cognitive abilities for learning, and can make studying on Sunday and performing well on a test on Monday much more difficult.
Myth No. 5: Marijuana is not addictive
Marijuana is addictive, with studies suggesting that nearly 9% of marijuana users will become addicted. Again, the risks are far greater for young people. Among people who begin using marijuana during adolescence, the rate of addiction climbs to 17%, and can be as high as 50% in daily users. Remember that addiction describes a pattern of continued use despite that use causing significant legal, social, or school and work problems. Users also may develop physical dependence, with a withdrawal syndrome that includes irritability, restlessness, insomnia, and appetite changes; these can last as long as 2 weeks.
Currently available forms of marijuana are much more potent than those that were studied and used in prior decades. On average, the potency of smoked marijuana has tripled, and there are concentrates (in oil form, for example) and hybrids with much higher potency still. More potent marijuana increases the high from even a small dose, and increases the likelihood of addiction and of other immediate and lingering complications of its use. So, parents who think they know what marijuana does to adolescents based on their own youthful experiences are significantly underestimating the risks.
When asking your patients explicitly about marijuana use, be curious and nonjudgmental, but also be frank and forthright about what is known about the risks associated with its use. Although the current legal and political changes around marijuana use may have given them the impression that marijuana use is safe, you want them to have the facts they need to make informed decisions. Even if you only discuss one of these myths with your patients, you will have equipped them with powerful information that they may use and share with their friends.
Dr. Swick is an attending psychiatrist in the division of child psychiatry at Massachusetts General Hospital, Boston, and director of the Parenting at a Challenging Time (PACT) Program at the Vernon Cancer Center at Newton (Mass.) Wellesley Hospital. Dr. Jellinek is professor of psychiatry and of pediatrics at Harvard Medical School, Boston. Email them at pdnews@frontlinemedcom.com.
The annual checkup has long provided an opportunity for early adolescents to learn about the risks of alcohol and drug use from a trusted source who may be less biased than parents, teachers, or police. Parents also turn to their child’s pediatrician for guidance on how to broach this important topic with their children, or they may come with concerns about their children’s use of drugs or alcohol.
Marijuana has become an increasingly complex topic, as its legal status has rapidly changed: It’s legal to purchase marijuana in four states (Alaska, Colorado, Oregon, and Washington, as well as the District of Columbia); it is decriminalized in 20 states and the District of Columbia for certain marijuana possession offenses; and it is legal to use medical marijuana in 23 states. As its legal status changes, attitudes about its use also have shifted, and its availability, form, and potency all have changed dramatically in just the past decade. Further, we ourselves may have mixed feelings about marijuana use based on our own experience as adolescents and sampling bias. We may have seen its low-level use and minimal effects in young or mature adults, or we may have seen substantial use of marijuana have a major deleterious impact on a friend or become a gateway drug for addiction to dangerous substances.
Before addressing marijuana use with adolescent patients and dealing with their potential skepticism concerning any harm, it is worth spending a little time looking in the mirror to consider your perspective on marijuana use and your response to disbelief.
According to the National Institute on Drug Abuse’s Monitoring the Future (MTF) survey, almost 12% of 8th graders, 27% of 10th graders, and 35% of 12th graders in the United States reported having used marijuana in the past year. Among the 12th graders in that 2014 survey, almost 20% were current users of marijuana and 6% were daily users. Many surveys, including the MTF, have demonstrated that attitudes of teenagers have shifted about marijuana’s dangerousness, with a steep and steady decline in the number of teenagers believing that regular marijuana use poses a risk to their health and well-being. In 2014, less than 40% of 12th graders in the MTF survey agreed that regular use of marijuana would pose a risk to their well-being, compared with a peak of almost 80% of 12th graders in the early 1990’s.
Pediatricians have an opportunity to change their patient’s thinking about marijuana. At the checkup when you routinely ask about alcohol and drug use, ask about marijuana use in particular. You might start by asking if they have heard their friends talking about marijuana? What have they heard? Are other kids using it? Have they ever seen anyone use it? Have their friends invited them to try? You should find out if they think it is safe or dangerous, and how it compares with cigarettes, alcohol, and other drugs on this score. Then you may be able to debunk some myths you hear from them.
Myth No. 1: Marijuana is medicine
Although 23 states allow the legal sale of marijuana for “medicinal purposes,” it is important to note that there are currently no Food and Drug Administration–approved indications for medical marijuana. There is modest evidence that the active compounds in marijuana (delta-9-tetra-hydrocannabinol [THC] and other cannabinoids) can be effective in the management of the muscle spasticity associated with multiple sclerosis, the treatment of nausea associated with chemotherapy, and increasing the appetite of patients with wasting due to AIDS, and there are FDA-approved synthetic cannabinoids that can be prescribed for these symptoms. It is also important to note that there is no evidence that THC or other cannabinoids are useful in the treatment of mood or anxiety symptoms, even though these are often used as reasons for seeking medicinal marijuana. Indeed, marijuana may cause or worsen several psychiatric problems.
Myth No. 2: Marijuana is safe
Although there is consensus that moderate marijuana use in adulthood poses only limited health risks (including the known risks of smoking), there is robust evidence that marijuana use during youth (through the early 20s) causes several serious and permanent effects on the developing brain. One 2012 study showed that for youth who are dependent on marijuana before they are 18 years, there is an 8-point drop in IQ in adulthood (Proc Natl Acad Sci USA. 2012 Oct 2;109[40]:E2657-64). This IQ drop persists even if they quit smoking, and does not occur for those who first become dependent on marijuana in adulthood. A 2015 study demonstrated that even for adolescents who are light smokers (one to two times weekly) with no evidence of marijuana dependence, there are significant abnormalities in the size and shape of their amygdala and nucleus accumbens, with associated changes in their motivation, decision making, attention, functional memory, and processing of emotions(J Neurosci. 2014 Apr 16;34[16]:5529-38). These abnormalities increase with increased frequency of use, and are not seen in those who begin smoking in adulthood (mid-20s and later).
Beyond these findings of cognitive deficits, evidence is growing that adolescent marijuana use is associated with several psychiatric illnesses, including depression and anxiety. There is especially strong evidence for a causal link between marijuana use and psychotic illnesses in (genetically) vulnerable young people. Any marijuana user can experience a brief psychotic reaction if the amount ingested or smoked is great enough, but for those young people who carry a specific variant of the gene for catechol-o-methyltransferase (COMT, an enzyme that degrades neurotransmitters), smoking marijuana in adolescence nearly triples their risk of developing schizophrenia in adulthood. For youth with a variant of the AKT gene (another enzyme affecting dopamine signaling in the brain), daily use of marijuana raises their risk of developing schizophrenia sevenfold. Clearly, marijuana can be the critical environmental trigger for schizophrenia in genetically vulnerable youth. Until we have a comprehensive knowledge of the relevant genes, and routinely check every patient’s complete genetic profile, it is reasonable to assume that any young person using marijuana is significantly increasing the risk of developing schizophrenia, a chronic and disabling condition.
Myth No. 3: Marijuana has no effect on driving
Marijuana intoxication significantly affects motor coordination, reaction time, and judgment, and multiple studies have demonstrated a direct relationship between blood THC concentration and impaired driving ability. A recent meta-analysis demonstrated that the risk of being in a car accident doubled after marijuana use (Drug Alcohol Depend. 2004 Feb 7;73[2]:109-19). These studies usually involved adults, and it is reasonable to assume that the risks may be more pronounced in adolescents, particularly ones who are new to driving or have other problems that could affect their attention or reaction time (such as attention-deficit/hyperactivity disorder). Beyond letting patients know about the increased risks of accidents, it may be worth reminding them that driving while intoxicated – even with legal use marijuana – is a criminal offense.
Myth No. 4: Marijuana has no effect on schoolwork
Aside from the risks of causing long-term cognitive changes and psychiatric problems that can affect school performance, the direct effects of marijuana intoxication can linger and affect school performance well after its use. The “high” from marijuana typically lasts from 1 to 3 hours, but the drug’s effects on higher-level cognitive processes (mediated by the neocortex and hippocampus) can last for days. So a teenager who smokes on Saturday night may have lingering impairment of motivation, the ability to shift attention, the ability to learn complex tasks, and working memory. These are all critical cognitive abilities for learning, and can make studying on Sunday and performing well on a test on Monday much more difficult.
Myth No. 5: Marijuana is not addictive
Marijuana is addictive, with studies suggesting that nearly 9% of marijuana users will become addicted. Again, the risks are far greater for young people. Among people who begin using marijuana during adolescence, the rate of addiction climbs to 17%, and can be as high as 50% in daily users. Remember that addiction describes a pattern of continued use despite that use causing significant legal, social, or school and work problems. Users also may develop physical dependence, with a withdrawal syndrome that includes irritability, restlessness, insomnia, and appetite changes; these can last as long as 2 weeks.
Currently available forms of marijuana are much more potent than those that were studied and used in prior decades. On average, the potency of smoked marijuana has tripled, and there are concentrates (in oil form, for example) and hybrids with much higher potency still. More potent marijuana increases the high from even a small dose, and increases the likelihood of addiction and of other immediate and lingering complications of its use. So, parents who think they know what marijuana does to adolescents based on their own youthful experiences are significantly underestimating the risks.
When asking your patients explicitly about marijuana use, be curious and nonjudgmental, but also be frank and forthright about what is known about the risks associated with its use. Although the current legal and political changes around marijuana use may have given them the impression that marijuana use is safe, you want them to have the facts they need to make informed decisions. Even if you only discuss one of these myths with your patients, you will have equipped them with powerful information that they may use and share with their friends.
Dr. Swick is an attending psychiatrist in the division of child psychiatry at Massachusetts General Hospital, Boston, and director of the Parenting at a Challenging Time (PACT) Program at the Vernon Cancer Center at Newton (Mass.) Wellesley Hospital. Dr. Jellinek is professor of psychiatry and of pediatrics at Harvard Medical School, Boston. Email them at pdnews@frontlinemedcom.com.
The annual checkup has long provided an opportunity for early adolescents to learn about the risks of alcohol and drug use from a trusted source who may be less biased than parents, teachers, or police. Parents also turn to their child’s pediatrician for guidance on how to broach this important topic with their children, or they may come with concerns about their children’s use of drugs or alcohol.
Marijuana has become an increasingly complex topic, as its legal status has rapidly changed: It’s legal to purchase marijuana in four states (Alaska, Colorado, Oregon, and Washington, as well as the District of Columbia); it is decriminalized in 20 states and the District of Columbia for certain marijuana possession offenses; and it is legal to use medical marijuana in 23 states. As its legal status changes, attitudes about its use also have shifted, and its availability, form, and potency all have changed dramatically in just the past decade. Further, we ourselves may have mixed feelings about marijuana use based on our own experience as adolescents and sampling bias. We may have seen its low-level use and minimal effects in young or mature adults, or we may have seen substantial use of marijuana have a major deleterious impact on a friend or become a gateway drug for addiction to dangerous substances.
Before addressing marijuana use with adolescent patients and dealing with their potential skepticism concerning any harm, it is worth spending a little time looking in the mirror to consider your perspective on marijuana use and your response to disbelief.
According to the National Institute on Drug Abuse’s Monitoring the Future (MTF) survey, almost 12% of 8th graders, 27% of 10th graders, and 35% of 12th graders in the United States reported having used marijuana in the past year. Among the 12th graders in that 2014 survey, almost 20% were current users of marijuana and 6% were daily users. Many surveys, including the MTF, have demonstrated that attitudes of teenagers have shifted about marijuana’s dangerousness, with a steep and steady decline in the number of teenagers believing that regular marijuana use poses a risk to their health and well-being. In 2014, less than 40% of 12th graders in the MTF survey agreed that regular use of marijuana would pose a risk to their well-being, compared with a peak of almost 80% of 12th graders in the early 1990’s.
Pediatricians have an opportunity to change their patient’s thinking about marijuana. At the checkup when you routinely ask about alcohol and drug use, ask about marijuana use in particular. You might start by asking if they have heard their friends talking about marijuana? What have they heard? Are other kids using it? Have they ever seen anyone use it? Have their friends invited them to try? You should find out if they think it is safe or dangerous, and how it compares with cigarettes, alcohol, and other drugs on this score. Then you may be able to debunk some myths you hear from them.
Myth No. 1: Marijuana is medicine
Although 23 states allow the legal sale of marijuana for “medicinal purposes,” it is important to note that there are currently no Food and Drug Administration–approved indications for medical marijuana. There is modest evidence that the active compounds in marijuana (delta-9-tetra-hydrocannabinol [THC] and other cannabinoids) can be effective in the management of the muscle spasticity associated with multiple sclerosis, the treatment of nausea associated with chemotherapy, and increasing the appetite of patients with wasting due to AIDS, and there are FDA-approved synthetic cannabinoids that can be prescribed for these symptoms. It is also important to note that there is no evidence that THC or other cannabinoids are useful in the treatment of mood or anxiety symptoms, even though these are often used as reasons for seeking medicinal marijuana. Indeed, marijuana may cause or worsen several psychiatric problems.
Myth No. 2: Marijuana is safe
Although there is consensus that moderate marijuana use in adulthood poses only limited health risks (including the known risks of smoking), there is robust evidence that marijuana use during youth (through the early 20s) causes several serious and permanent effects on the developing brain. One 2012 study showed that for youth who are dependent on marijuana before they are 18 years, there is an 8-point drop in IQ in adulthood (Proc Natl Acad Sci USA. 2012 Oct 2;109[40]:E2657-64). This IQ drop persists even if they quit smoking, and does not occur for those who first become dependent on marijuana in adulthood. A 2015 study demonstrated that even for adolescents who are light smokers (one to two times weekly) with no evidence of marijuana dependence, there are significant abnormalities in the size and shape of their amygdala and nucleus accumbens, with associated changes in their motivation, decision making, attention, functional memory, and processing of emotions(J Neurosci. 2014 Apr 16;34[16]:5529-38). These abnormalities increase with increased frequency of use, and are not seen in those who begin smoking in adulthood (mid-20s and later).
Beyond these findings of cognitive deficits, evidence is growing that adolescent marijuana use is associated with several psychiatric illnesses, including depression and anxiety. There is especially strong evidence for a causal link between marijuana use and psychotic illnesses in (genetically) vulnerable young people. Any marijuana user can experience a brief psychotic reaction if the amount ingested or smoked is great enough, but for those young people who carry a specific variant of the gene for catechol-o-methyltransferase (COMT, an enzyme that degrades neurotransmitters), smoking marijuana in adolescence nearly triples their risk of developing schizophrenia in adulthood. For youth with a variant of the AKT gene (another enzyme affecting dopamine signaling in the brain), daily use of marijuana raises their risk of developing schizophrenia sevenfold. Clearly, marijuana can be the critical environmental trigger for schizophrenia in genetically vulnerable youth. Until we have a comprehensive knowledge of the relevant genes, and routinely check every patient’s complete genetic profile, it is reasonable to assume that any young person using marijuana is significantly increasing the risk of developing schizophrenia, a chronic and disabling condition.
Myth No. 3: Marijuana has no effect on driving
Marijuana intoxication significantly affects motor coordination, reaction time, and judgment, and multiple studies have demonstrated a direct relationship between blood THC concentration and impaired driving ability. A recent meta-analysis demonstrated that the risk of being in a car accident doubled after marijuana use (Drug Alcohol Depend. 2004 Feb 7;73[2]:109-19). These studies usually involved adults, and it is reasonable to assume that the risks may be more pronounced in adolescents, particularly ones who are new to driving or have other problems that could affect their attention or reaction time (such as attention-deficit/hyperactivity disorder). Beyond letting patients know about the increased risks of accidents, it may be worth reminding them that driving while intoxicated – even with legal use marijuana – is a criminal offense.
Myth No. 4: Marijuana has no effect on schoolwork
Aside from the risks of causing long-term cognitive changes and psychiatric problems that can affect school performance, the direct effects of marijuana intoxication can linger and affect school performance well after its use. The “high” from marijuana typically lasts from 1 to 3 hours, but the drug’s effects on higher-level cognitive processes (mediated by the neocortex and hippocampus) can last for days. So a teenager who smokes on Saturday night may have lingering impairment of motivation, the ability to shift attention, the ability to learn complex tasks, and working memory. These are all critical cognitive abilities for learning, and can make studying on Sunday and performing well on a test on Monday much more difficult.
Myth No. 5: Marijuana is not addictive
Marijuana is addictive, with studies suggesting that nearly 9% of marijuana users will become addicted. Again, the risks are far greater for young people. Among people who begin using marijuana during adolescence, the rate of addiction climbs to 17%, and can be as high as 50% in daily users. Remember that addiction describes a pattern of continued use despite that use causing significant legal, social, or school and work problems. Users also may develop physical dependence, with a withdrawal syndrome that includes irritability, restlessness, insomnia, and appetite changes; these can last as long as 2 weeks.
Currently available forms of marijuana are much more potent than those that were studied and used in prior decades. On average, the potency of smoked marijuana has tripled, and there are concentrates (in oil form, for example) and hybrids with much higher potency still. More potent marijuana increases the high from even a small dose, and increases the likelihood of addiction and of other immediate and lingering complications of its use. So, parents who think they know what marijuana does to adolescents based on their own youthful experiences are significantly underestimating the risks.
When asking your patients explicitly about marijuana use, be curious and nonjudgmental, but also be frank and forthright about what is known about the risks associated with its use. Although the current legal and political changes around marijuana use may have given them the impression that marijuana use is safe, you want them to have the facts they need to make informed decisions. Even if you only discuss one of these myths with your patients, you will have equipped them with powerful information that they may use and share with their friends.
Dr. Swick is an attending psychiatrist in the division of child psychiatry at Massachusetts General Hospital, Boston, and director of the Parenting at a Challenging Time (PACT) Program at the Vernon Cancer Center at Newton (Mass.) Wellesley Hospital. Dr. Jellinek is professor of psychiatry and of pediatrics at Harvard Medical School, Boston. Email them at pdnews@frontlinemedcom.com.
Age, But Not Sex, Is Associated With the Efficacy of IV Migraine Treatment
Although migraine prevalence is associated with sex, sex is not associated with response to IV migraine medication, according to research published online ahead of print October 21 in Headache. Age, however, is inversely associated with the efficacy of IV migraine medication and directly associated with the risk of adverse events.
An Analysis of Three Migraine Trials
Benjamin W. Friedman, MD, Associate Professor of Emergency Medicine at Albert Einstein College of Medicine in Bronx, New York, and colleagues examined data gathered during three emergency-department-based randomized comparative efficacy trials of various migraine medications to determine whether sex and age are associated with short-term headache relief, sustained headache freedom, or adverse medication effects. The medications studied in the trials included metoclopramide, an antiemetic dopamine antagonist; ketorolac, a nonsteroidal anti-inflammatory drug; dexamethasone, a corticosteroid; and valproate, an antiepileptic drug. In each of the clinical trials, patients presenting to an emergency department with acute migraine were randomized to treatment with one or a combination of the IV drugs.
Eligible patients had an acute headache that fulfilled all International Classification of Headache Disorders criteria for migraine without aura. Patients were not excluded for prolonged duration of headache, however. Nor were patients required to have had more than one similar previous headache. Dr. Friedman and colleagues assessed pain levels and medication side effects at baseline, at one and two hours after medication administration, and by telephone at 24–48 hours after discharge from the emergency department. Patients described their pain as none, mild, moderate, or severe.
The researchers chose short-term headache relief (ie, a headache level of mild or none within one hour of treatment) and sustained headache freedom (ie, a reported headache level of none in the emergency department and a period of headache freedom of at least 24 hours after discharge) as their efficacy end points. The investigators’ third outcome was side effects.
For the primary analysis, Dr. Friedman and colleagues used the population’s median age to separate participants into an older and a younger group. In a secondary analysis, they considered age as continuous data.
Nausea Was More Common in Older Patients
The three original studies included 884 participants. The investigators found no differences between men and women in terms of age, race, duration of headache, or presence of aura. Compared with men, women were more likely to be nauseated (56% vs 41%) and to report severe pain at baseline (69% vs 59%). Similarly, patients age 36 or older were more likely to be nauseated (57% vs 50%) and to report severe pain at baseline (72% vs 64%) than patients younger than 36. “We are the first to report [this finding],” said Dr. Friedman.
Men and women were approximately equally likely to report short-term and sustained headache improvements and adverse events. Patients age 36 or older, however, were less likely than younger patients to respond favorably to headache medication and more likely to have adverse events. Most of the difference in efficacy outcomes between older and younger patients can be explained by differential response to metoclopramide regimens, according to the authors.
Bivariate analyses performed for various medication types indicated that adults age 36 or older were less likely to respond to combinations of metoclopramide and diphenhydramine. Age was not associated with the efficacy of ketorolac or valproate, however. “Because the bulk of our data comes from patients who received metoclopramide, these data do not necessarily translate to other medication classes,” said Dr. Friedman. Although adults older than 35 may be less likely to respond to metoclopramide regimens, it is uncertain that they are more likely to respond to alternate treatment regimens, he added. “Therefore, one should not reject metoclopramide in adults older than 35 based solely on these data.”
The higher rate of adverse events in older patients primarily resulted from a disparity in response to dexamethasone. Adults older than 35 who received dexamethasone had an adverse event rate of more than 50%. “Because the benefit of dexamethasone for patients with acute episodic migraine is relatively modest, clinicians may wish to avoid administering this medication to these patients,” said Dr. Friedman.
After the investigators performed multivariate logistic regression modeling, they found that sex did not meaningfully influence the association between age and the study outcomes. Likewise, age did not influence the lack of association between sex and the study outcomes.
Bioavailability May Decline With Age
The reasons for age-related differences in the efficacy of IV migraine medication are a matter of debate. One hypothesis is that age-related alterations in pharmacodynamics may affect the drugs’ bioavailability, said the authors. In addition, previous research indicates that endogenous pain modulation declines with age and affects the middle-aged and the elderly. Another potential explanation involves the psychologic response to investigational medicine. In outpatient triptan trials, middle-aged adults with migraine were less likely to respond to oral placebo than younger adults were. “Thus, these data may not represent a difference in true efficacy at all, but rather a diminished expectation of treatment benefit,” said Dr. Friedman.
“Other investigators reported that women experience migraine recurrence after successful treatment with oral triptans more frequently than men,” said Dr. Friedman. “We were not able to replicate this latter finding in our analysis of sustained headache response, which incorporates the potential for headache recurrence.
“We were unable to find other clinical data that addressed the association between age and response to acute parenteral migraine medication,” he continued. “As with sex, other investigators have reported that middle-aged adults were more likely than younger adults to report recurrence of headache after using an oral triptan. This finding was not supported in our assessment of sustained outcomes.”
The authors acknowledged several limitations of their study, including a gender imbalance in the patient population that resulted from the higher prevalence of migraine among women. Although the imbalance decreased the power of the sex analysis, the data appear sufficiently robust to detect meaningful differences, said Dr. Friedman.
“Future work should investigate sex- and age-based response to other parenteral migraine medications, including migraine-specific agents such as sumatriptan and dihydroergotamine,” Dr. Friedman concluded.
—Erik Greb
Suggested Reading
Friedman BW, Cisewski DH, Holden L, et al. Age but not sex is associated with efficacy and adverse events following administration of intravenous migraine medication: an analysis of a clinical trial database. Headache. 2015 Oct 21 [Epub ahead of print].
Although migraine prevalence is associated with sex, sex is not associated with response to IV migraine medication, according to research published online ahead of print October 21 in Headache. Age, however, is inversely associated with the efficacy of IV migraine medication and directly associated with the risk of adverse events.
An Analysis of Three Migraine Trials
Benjamin W. Friedman, MD, Associate Professor of Emergency Medicine at Albert Einstein College of Medicine in Bronx, New York, and colleagues examined data gathered during three emergency-department-based randomized comparative efficacy trials of various migraine medications to determine whether sex and age are associated with short-term headache relief, sustained headache freedom, or adverse medication effects. The medications studied in the trials included metoclopramide, an antiemetic dopamine antagonist; ketorolac, a nonsteroidal anti-inflammatory drug; dexamethasone, a corticosteroid; and valproate, an antiepileptic drug. In each of the clinical trials, patients presenting to an emergency department with acute migraine were randomized to treatment with one or a combination of the IV drugs.
Eligible patients had an acute headache that fulfilled all International Classification of Headache Disorders criteria for migraine without aura. Patients were not excluded for prolonged duration of headache, however. Nor were patients required to have had more than one similar previous headache. Dr. Friedman and colleagues assessed pain levels and medication side effects at baseline, at one and two hours after medication administration, and by telephone at 24–48 hours after discharge from the emergency department. Patients described their pain as none, mild, moderate, or severe.
The researchers chose short-term headache relief (ie, a headache level of mild or none within one hour of treatment) and sustained headache freedom (ie, a reported headache level of none in the emergency department and a period of headache freedom of at least 24 hours after discharge) as their efficacy end points. The investigators’ third outcome was side effects.
For the primary analysis, Dr. Friedman and colleagues used the population’s median age to separate participants into an older and a younger group. In a secondary analysis, they considered age as continuous data.
Nausea Was More Common in Older Patients
The three original studies included 884 participants. The investigators found no differences between men and women in terms of age, race, duration of headache, or presence of aura. Compared with men, women were more likely to be nauseated (56% vs 41%) and to report severe pain at baseline (69% vs 59%). Similarly, patients age 36 or older were more likely to be nauseated (57% vs 50%) and to report severe pain at baseline (72% vs 64%) than patients younger than 36. “We are the first to report [this finding],” said Dr. Friedman.
Men and women were approximately equally likely to report short-term and sustained headache improvements and adverse events. Patients age 36 or older, however, were less likely than younger patients to respond favorably to headache medication and more likely to have adverse events. Most of the difference in efficacy outcomes between older and younger patients can be explained by differential response to metoclopramide regimens, according to the authors.
Bivariate analyses performed for various medication types indicated that adults age 36 or older were less likely to respond to combinations of metoclopramide and diphenhydramine. Age was not associated with the efficacy of ketorolac or valproate, however. “Because the bulk of our data comes from patients who received metoclopramide, these data do not necessarily translate to other medication classes,” said Dr. Friedman. Although adults older than 35 may be less likely to respond to metoclopramide regimens, it is uncertain that they are more likely to respond to alternate treatment regimens, he added. “Therefore, one should not reject metoclopramide in adults older than 35 based solely on these data.”
The higher rate of adverse events in older patients primarily resulted from a disparity in response to dexamethasone. Adults older than 35 who received dexamethasone had an adverse event rate of more than 50%. “Because the benefit of dexamethasone for patients with acute episodic migraine is relatively modest, clinicians may wish to avoid administering this medication to these patients,” said Dr. Friedman.
After the investigators performed multivariate logistic regression modeling, they found that sex did not meaningfully influence the association between age and the study outcomes. Likewise, age did not influence the lack of association between sex and the study outcomes.
Bioavailability May Decline With Age
The reasons for age-related differences in the efficacy of IV migraine medication are a matter of debate. One hypothesis is that age-related alterations in pharmacodynamics may affect the drugs’ bioavailability, said the authors. In addition, previous research indicates that endogenous pain modulation declines with age and affects the middle-aged and the elderly. Another potential explanation involves the psychologic response to investigational medicine. In outpatient triptan trials, middle-aged adults with migraine were less likely to respond to oral placebo than younger adults were. “Thus, these data may not represent a difference in true efficacy at all, but rather a diminished expectation of treatment benefit,” said Dr. Friedman.
“Other investigators reported that women experience migraine recurrence after successful treatment with oral triptans more frequently than men,” said Dr. Friedman. “We were not able to replicate this latter finding in our analysis of sustained headache response, which incorporates the potential for headache recurrence.
“We were unable to find other clinical data that addressed the association between age and response to acute parenteral migraine medication,” he continued. “As with sex, other investigators have reported that middle-aged adults were more likely than younger adults to report recurrence of headache after using an oral triptan. This finding was not supported in our assessment of sustained outcomes.”
The authors acknowledged several limitations of their study, including a gender imbalance in the patient population that resulted from the higher prevalence of migraine among women. Although the imbalance decreased the power of the sex analysis, the data appear sufficiently robust to detect meaningful differences, said Dr. Friedman.
“Future work should investigate sex- and age-based response to other parenteral migraine medications, including migraine-specific agents such as sumatriptan and dihydroergotamine,” Dr. Friedman concluded.
—Erik Greb
Although migraine prevalence is associated with sex, sex is not associated with response to IV migraine medication, according to research published online ahead of print October 21 in Headache. Age, however, is inversely associated with the efficacy of IV migraine medication and directly associated with the risk of adverse events.
An Analysis of Three Migraine Trials
Benjamin W. Friedman, MD, Associate Professor of Emergency Medicine at Albert Einstein College of Medicine in Bronx, New York, and colleagues examined data gathered during three emergency-department-based randomized comparative efficacy trials of various migraine medications to determine whether sex and age are associated with short-term headache relief, sustained headache freedom, or adverse medication effects. The medications studied in the trials included metoclopramide, an antiemetic dopamine antagonist; ketorolac, a nonsteroidal anti-inflammatory drug; dexamethasone, a corticosteroid; and valproate, an antiepileptic drug. In each of the clinical trials, patients presenting to an emergency department with acute migraine were randomized to treatment with one or a combination of the IV drugs.
Eligible patients had an acute headache that fulfilled all International Classification of Headache Disorders criteria for migraine without aura. Patients were not excluded for prolonged duration of headache, however. Nor were patients required to have had more than one similar previous headache. Dr. Friedman and colleagues assessed pain levels and medication side effects at baseline, at one and two hours after medication administration, and by telephone at 24–48 hours after discharge from the emergency department. Patients described their pain as none, mild, moderate, or severe.
The researchers chose short-term headache relief (ie, a headache level of mild or none within one hour of treatment) and sustained headache freedom (ie, a reported headache level of none in the emergency department and a period of headache freedom of at least 24 hours after discharge) as their efficacy end points. The investigators’ third outcome was side effects.
For the primary analysis, Dr. Friedman and colleagues used the population’s median age to separate participants into an older and a younger group. In a secondary analysis, they considered age as continuous data.
Nausea Was More Common in Older Patients
The three original studies included 884 participants. The investigators found no differences between men and women in terms of age, race, duration of headache, or presence of aura. Compared with men, women were more likely to be nauseated (56% vs 41%) and to report severe pain at baseline (69% vs 59%). Similarly, patients age 36 or older were more likely to be nauseated (57% vs 50%) and to report severe pain at baseline (72% vs 64%) than patients younger than 36. “We are the first to report [this finding],” said Dr. Friedman.
Men and women were approximately equally likely to report short-term and sustained headache improvements and adverse events. Patients age 36 or older, however, were less likely than younger patients to respond favorably to headache medication and more likely to have adverse events. Most of the difference in efficacy outcomes between older and younger patients can be explained by differential response to metoclopramide regimens, according to the authors.
Bivariate analyses performed for various medication types indicated that adults age 36 or older were less likely to respond to combinations of metoclopramide and diphenhydramine. Age was not associated with the efficacy of ketorolac or valproate, however. “Because the bulk of our data comes from patients who received metoclopramide, these data do not necessarily translate to other medication classes,” said Dr. Friedman. Although adults older than 35 may be less likely to respond to metoclopramide regimens, it is uncertain that they are more likely to respond to alternate treatment regimens, he added. “Therefore, one should not reject metoclopramide in adults older than 35 based solely on these data.”
The higher rate of adverse events in older patients primarily resulted from a disparity in response to dexamethasone. Adults older than 35 who received dexamethasone had an adverse event rate of more than 50%. “Because the benefit of dexamethasone for patients with acute episodic migraine is relatively modest, clinicians may wish to avoid administering this medication to these patients,” said Dr. Friedman.
After the investigators performed multivariate logistic regression modeling, they found that sex did not meaningfully influence the association between age and the study outcomes. Likewise, age did not influence the lack of association between sex and the study outcomes.
Bioavailability May Decline With Age
The reasons for age-related differences in the efficacy of IV migraine medication are a matter of debate. One hypothesis is that age-related alterations in pharmacodynamics may affect the drugs’ bioavailability, said the authors. In addition, previous research indicates that endogenous pain modulation declines with age and affects the middle-aged and the elderly. Another potential explanation involves the psychologic response to investigational medicine. In outpatient triptan trials, middle-aged adults with migraine were less likely to respond to oral placebo than younger adults were. “Thus, these data may not represent a difference in true efficacy at all, but rather a diminished expectation of treatment benefit,” said Dr. Friedman.
“Other investigators reported that women experience migraine recurrence after successful treatment with oral triptans more frequently than men,” said Dr. Friedman. “We were not able to replicate this latter finding in our analysis of sustained headache response, which incorporates the potential for headache recurrence.
“We were unable to find other clinical data that addressed the association between age and response to acute parenteral migraine medication,” he continued. “As with sex, other investigators have reported that middle-aged adults were more likely than younger adults to report recurrence of headache after using an oral triptan. This finding was not supported in our assessment of sustained outcomes.”
The authors acknowledged several limitations of their study, including a gender imbalance in the patient population that resulted from the higher prevalence of migraine among women. Although the imbalance decreased the power of the sex analysis, the data appear sufficiently robust to detect meaningful differences, said Dr. Friedman.
“Future work should investigate sex- and age-based response to other parenteral migraine medications, including migraine-specific agents such as sumatriptan and dihydroergotamine,” Dr. Friedman concluded.
—Erik Greb
Suggested Reading
Friedman BW, Cisewski DH, Holden L, et al. Age but not sex is associated with efficacy and adverse events following administration of intravenous migraine medication: an analysis of a clinical trial database. Headache. 2015 Oct 21 [Epub ahead of print].
Suggested Reading
Friedman BW, Cisewski DH, Holden L, et al. Age but not sex is associated with efficacy and adverse events following administration of intravenous migraine medication: an analysis of a clinical trial database. Headache. 2015 Oct 21 [Epub ahead of print].
Pisa Syndrome May Be a Relatively Common Complication in Parkinson’s Disease
Pisa syndrome may be a relatively frequent and often disabling complication in Parkinson’s disease, especially in the advanced disease stages, according to research published in the November 17 issue of Neurology.
Variables associated with Pisa syndrome include Hoehn and Yahr stage, ongoing combined treatment with levodopa and dopamine agonist, and veering gait, said Michele Tinazzi, MD, PhD, Associate Professor of Neurology at the University of Verona, Italy, and colleagues.
Pisa syndrome, which is characterized by lateral trunk flexion, has been described in patients with dementia and other neurodegenerative diseases. It was first described as a side effect of antipsychotic treatment and also has been associated with medications such as antiemetics, antidepressants, central cholinesterase inhibitors, and lithium carbonate. Pisa syndrome’s pathophysiologic explanation, however, is uncertain, and case series have produced conflicting findings, the researchers said.
A Cross-Sectional Study of Patients With Parkinson’s Disease
To assess the prevalence of Pisa syndrome in patients with Parkinson’s disease and the association between Pisa syndrome and demographic and clinical variables, Dr. Tinazzi and colleagues conducted a cross-sectional study of consecutive outpatients with Parkinson’s disease who attended 21 tertiary movement disorders centers in Italy.
The investigators enrolled patients between February 2012 and July 2013. Patients diagnosed with Pisa syndrome (ie, lateral trunk deviation of 10° or more that was almost completely reverted by passive mobilization or supine positioning) completed an ad hoc questionnaire and neurologic examination. The researchers diagnosed Parkinson’s disease according to the United Kingdom Parkinson’s Disease Society Brain Bank criteria and excluded patients who had other postural deformities, concomitant neurologic diseases known to affect posture, history of major spinal surgery or muscle or skeletal disease, treatment with drugs that can induce Pisa syndrome in the six months before enrollment, and clinical features that were consistent with a diagnosis of atypical parkinsonism.
A neurologist at each center recorded patients’ sex, age, age at Parkinson’s disease onset, BMI, disease duration, Parkinson’s disease phenotype (ie, rigid-akinetic, tremor-dominant, or mixed type), laterality of motor symptoms at disease onset, latency between disease onset and the start of antiparkinsonian therapy, and pharmacologic treatment at disease onset and at their latest visit.
The researchers also evaluated falls in the previous month; comorbidities; quality of life, as assessed by Parkinson’s Disease Questionnaire–8; and disease severity, as assessed by Unified Parkinson’s Disease Rating Scale, Parts I–IV. Clinical asymmetry and Hoehn and Yahr scale staging also were assessed. Investigators measured trunk deviation using a wall goniometer. Patients underwent a spine x-ray to disclose orthopedic conditions that could lead to lateral bending of the trunk.
Pisa Syndrome Was Associated With Age
Of the 1,631 patients who met the eligibility criteria, 143 patients fulfilled the diagnostic criteria for Pisa syndrome, representing a prevalence of 8.8%. Trunk flexion ranged from 10° to 50°, with an average of 17°. Pisa syndrome appeared on average seven years after the onset of Parkinson’s disease, and the patients had had Pisa syndrome for a mean of 2.6 years.
The investigators found that patients with Pisa syndrome were older and had lower BMI, a significantly longer disease duration, more severe disease, and worse quality of life, compared with patients who did not have Pisa syndrome. In addition, patients with Pisa syndrome had higher levodopa equivalent daily dose. Osteoporosis, arthrosis, veering gait, and falls were more common in the Pisa-syndrome group, compared with the group without Pisa syndrome. Multivariate logistic regression analysis confirmed that Hoehn and Yahr stage, ongoing antiparkinsonian treatment, associated medical conditions, and veering gait were associated with Pisa syndrome.
Most patients were aware of their leaning posture, and half adopted a head compensation to correct their visual alignment. Those with more severe Pisa syndrome (ie, trunk flexion of 20° or greater) were not significantly different from those with mild Pisa syndrome, in terms of demographic or clinical variables.
“The association of poor quality of life with Pisa syndrome in our cohort supports its clinical effect as motor manifestation of Parkinson’s disease; however, this was not confirmed by multivariate logistic regression analysis, suggesting that Pisa syndrome might not be the principal determinant of poor quality of life, but other factors associated with longer disease duration are also contributing,” the researchers said.
The study did not confirm a finding of previous studies that patients with Pisa syndrome lean away from their dominant Parkinson’s disease side.
These results may inform future studies that investigate the pathophysiologic mechanisms of Pisa syndrome in Parkinson’s disease and help identify “at-risk patients who may benefit from tailored therapeutic strategies,” said Dr. Tinazzi and colleagues.
“Early detection and treatment of Pisa syndrome may prevent fixed, irreversible deformities, thereby avoiding complications that may arise from such a disabling condition,” concluded the researchers.
—Jake Remaly
Suggested Reading
Tinazzi M, Fasano A, Geroin C, et al. Pisa syndrome in Parkinson disease: An observational multicenter Italian study. Neurology. 2015;85(20):1769-1779.
Pisa syndrome may be a relatively frequent and often disabling complication in Parkinson’s disease, especially in the advanced disease stages, according to research published in the November 17 issue of Neurology.
Variables associated with Pisa syndrome include Hoehn and Yahr stage, ongoing combined treatment with levodopa and dopamine agonist, and veering gait, said Michele Tinazzi, MD, PhD, Associate Professor of Neurology at the University of Verona, Italy, and colleagues.
Pisa syndrome, which is characterized by lateral trunk flexion, has been described in patients with dementia and other neurodegenerative diseases. It was first described as a side effect of antipsychotic treatment and also has been associated with medications such as antiemetics, antidepressants, central cholinesterase inhibitors, and lithium carbonate. Pisa syndrome’s pathophysiologic explanation, however, is uncertain, and case series have produced conflicting findings, the researchers said.
A Cross-Sectional Study of Patients With Parkinson’s Disease
To assess the prevalence of Pisa syndrome in patients with Parkinson’s disease and the association between Pisa syndrome and demographic and clinical variables, Dr. Tinazzi and colleagues conducted a cross-sectional study of consecutive outpatients with Parkinson’s disease who attended 21 tertiary movement disorders centers in Italy.
The investigators enrolled patients between February 2012 and July 2013. Patients diagnosed with Pisa syndrome (ie, lateral trunk deviation of 10° or more that was almost completely reverted by passive mobilization or supine positioning) completed an ad hoc questionnaire and neurologic examination. The researchers diagnosed Parkinson’s disease according to the United Kingdom Parkinson’s Disease Society Brain Bank criteria and excluded patients who had other postural deformities, concomitant neurologic diseases known to affect posture, history of major spinal surgery or muscle or skeletal disease, treatment with drugs that can induce Pisa syndrome in the six months before enrollment, and clinical features that were consistent with a diagnosis of atypical parkinsonism.
A neurologist at each center recorded patients’ sex, age, age at Parkinson’s disease onset, BMI, disease duration, Parkinson’s disease phenotype (ie, rigid-akinetic, tremor-dominant, or mixed type), laterality of motor symptoms at disease onset, latency between disease onset and the start of antiparkinsonian therapy, and pharmacologic treatment at disease onset and at their latest visit.
The researchers also evaluated falls in the previous month; comorbidities; quality of life, as assessed by Parkinson’s Disease Questionnaire–8; and disease severity, as assessed by Unified Parkinson’s Disease Rating Scale, Parts I–IV. Clinical asymmetry and Hoehn and Yahr scale staging also were assessed. Investigators measured trunk deviation using a wall goniometer. Patients underwent a spine x-ray to disclose orthopedic conditions that could lead to lateral bending of the trunk.
Pisa Syndrome Was Associated With Age
Of the 1,631 patients who met the eligibility criteria, 143 patients fulfilled the diagnostic criteria for Pisa syndrome, representing a prevalence of 8.8%. Trunk flexion ranged from 10° to 50°, with an average of 17°. Pisa syndrome appeared on average seven years after the onset of Parkinson’s disease, and the patients had had Pisa syndrome for a mean of 2.6 years.
The investigators found that patients with Pisa syndrome were older and had lower BMI, a significantly longer disease duration, more severe disease, and worse quality of life, compared with patients who did not have Pisa syndrome. In addition, patients with Pisa syndrome had higher levodopa equivalent daily dose. Osteoporosis, arthrosis, veering gait, and falls were more common in the Pisa-syndrome group, compared with the group without Pisa syndrome. Multivariate logistic regression analysis confirmed that Hoehn and Yahr stage, ongoing antiparkinsonian treatment, associated medical conditions, and veering gait were associated with Pisa syndrome.
Most patients were aware of their leaning posture, and half adopted a head compensation to correct their visual alignment. Those with more severe Pisa syndrome (ie, trunk flexion of 20° or greater) were not significantly different from those with mild Pisa syndrome, in terms of demographic or clinical variables.
“The association of poor quality of life with Pisa syndrome in our cohort supports its clinical effect as motor manifestation of Parkinson’s disease; however, this was not confirmed by multivariate logistic regression analysis, suggesting that Pisa syndrome might not be the principal determinant of poor quality of life, but other factors associated with longer disease duration are also contributing,” the researchers said.
The study did not confirm a finding of previous studies that patients with Pisa syndrome lean away from their dominant Parkinson’s disease side.
These results may inform future studies that investigate the pathophysiologic mechanisms of Pisa syndrome in Parkinson’s disease and help identify “at-risk patients who may benefit from tailored therapeutic strategies,” said Dr. Tinazzi and colleagues.
“Early detection and treatment of Pisa syndrome may prevent fixed, irreversible deformities, thereby avoiding complications that may arise from such a disabling condition,” concluded the researchers.
—Jake Remaly
Pisa syndrome may be a relatively frequent and often disabling complication in Parkinson’s disease, especially in the advanced disease stages, according to research published in the November 17 issue of Neurology.
Variables associated with Pisa syndrome include Hoehn and Yahr stage, ongoing combined treatment with levodopa and dopamine agonist, and veering gait, said Michele Tinazzi, MD, PhD, Associate Professor of Neurology at the University of Verona, Italy, and colleagues.
Pisa syndrome, which is characterized by lateral trunk flexion, has been described in patients with dementia and other neurodegenerative diseases. It was first described as a side effect of antipsychotic treatment and also has been associated with medications such as antiemetics, antidepressants, central cholinesterase inhibitors, and lithium carbonate. Pisa syndrome’s pathophysiologic explanation, however, is uncertain, and case series have produced conflicting findings, the researchers said.
A Cross-Sectional Study of Patients With Parkinson’s Disease
To assess the prevalence of Pisa syndrome in patients with Parkinson’s disease and the association between Pisa syndrome and demographic and clinical variables, Dr. Tinazzi and colleagues conducted a cross-sectional study of consecutive outpatients with Parkinson’s disease who attended 21 tertiary movement disorders centers in Italy.
The investigators enrolled patients between February 2012 and July 2013. Patients diagnosed with Pisa syndrome (ie, lateral trunk deviation of 10° or more that was almost completely reverted by passive mobilization or supine positioning) completed an ad hoc questionnaire and neurologic examination. The researchers diagnosed Parkinson’s disease according to the United Kingdom Parkinson’s Disease Society Brain Bank criteria and excluded patients who had other postural deformities, concomitant neurologic diseases known to affect posture, history of major spinal surgery or muscle or skeletal disease, treatment with drugs that can induce Pisa syndrome in the six months before enrollment, and clinical features that were consistent with a diagnosis of atypical parkinsonism.
A neurologist at each center recorded patients’ sex, age, age at Parkinson’s disease onset, BMI, disease duration, Parkinson’s disease phenotype (ie, rigid-akinetic, tremor-dominant, or mixed type), laterality of motor symptoms at disease onset, latency between disease onset and the start of antiparkinsonian therapy, and pharmacologic treatment at disease onset and at their latest visit.
The researchers also evaluated falls in the previous month; comorbidities; quality of life, as assessed by Parkinson’s Disease Questionnaire–8; and disease severity, as assessed by Unified Parkinson’s Disease Rating Scale, Parts I–IV. Clinical asymmetry and Hoehn and Yahr scale staging also were assessed. Investigators measured trunk deviation using a wall goniometer. Patients underwent a spine x-ray to disclose orthopedic conditions that could lead to lateral bending of the trunk.
Pisa Syndrome Was Associated With Age
Of the 1,631 patients who met the eligibility criteria, 143 patients fulfilled the diagnostic criteria for Pisa syndrome, representing a prevalence of 8.8%. Trunk flexion ranged from 10° to 50°, with an average of 17°. Pisa syndrome appeared on average seven years after the onset of Parkinson’s disease, and the patients had had Pisa syndrome for a mean of 2.6 years.
The investigators found that patients with Pisa syndrome were older and had lower BMI, a significantly longer disease duration, more severe disease, and worse quality of life, compared with patients who did not have Pisa syndrome. In addition, patients with Pisa syndrome had higher levodopa equivalent daily dose. Osteoporosis, arthrosis, veering gait, and falls were more common in the Pisa-syndrome group, compared with the group without Pisa syndrome. Multivariate logistic regression analysis confirmed that Hoehn and Yahr stage, ongoing antiparkinsonian treatment, associated medical conditions, and veering gait were associated with Pisa syndrome.
Most patients were aware of their leaning posture, and half adopted a head compensation to correct their visual alignment. Those with more severe Pisa syndrome (ie, trunk flexion of 20° or greater) were not significantly different from those with mild Pisa syndrome, in terms of demographic or clinical variables.
“The association of poor quality of life with Pisa syndrome in our cohort supports its clinical effect as motor manifestation of Parkinson’s disease; however, this was not confirmed by multivariate logistic regression analysis, suggesting that Pisa syndrome might not be the principal determinant of poor quality of life, but other factors associated with longer disease duration are also contributing,” the researchers said.
The study did not confirm a finding of previous studies that patients with Pisa syndrome lean away from their dominant Parkinson’s disease side.
These results may inform future studies that investigate the pathophysiologic mechanisms of Pisa syndrome in Parkinson’s disease and help identify “at-risk patients who may benefit from tailored therapeutic strategies,” said Dr. Tinazzi and colleagues.
“Early detection and treatment of Pisa syndrome may prevent fixed, irreversible deformities, thereby avoiding complications that may arise from such a disabling condition,” concluded the researchers.
—Jake Remaly
Suggested Reading
Tinazzi M, Fasano A, Geroin C, et al. Pisa syndrome in Parkinson disease: An observational multicenter Italian study. Neurology. 2015;85(20):1769-1779.
Suggested Reading
Tinazzi M, Fasano A, Geroin C, et al. Pisa syndrome in Parkinson disease: An observational multicenter Italian study. Neurology. 2015;85(20):1769-1779.
Compartment Syndrome in Children: Diagnosis and Management
Compartment syndrome (CS) is one of the true orthopedic emergencies. Identifying the high-risk patient, making a prompt diagnosis, and initiating effective treatment are the crucial steps in avoiding a poor outcome. A physician’s inability to communicate with young children can interfere with diagnosing CS in a timely fashion. Many young patients in hospitals are admitted to pediatric floors where routine orthopedic care is not the norm and staff are unfamiliar with the signs and symptoms of evolving CS. As orthopedic surgeons are often involved in caring for these patients, they should be aware of the aspects of CS that are unique to children and should be able to identify patients who are at risk and would benefit from close monitoring. In addition, given the consequences of late diagnosis, early diagnosis is important from a medicolegal standpoint. Only 44% of cases of adult and pediatric CS are decided in favor of treating physicians, compared with 75% of cases in other orthopedic malpractice claims.1,2
Risk Factors for Posttraumatic Compartment Syndrome
Supracondylar Humeral Fracture
CS is a well-described complication of this injury. CS develops in 0.1% to 0.3% of children who present with supracondylar humeral fracture.3,4 Casted elbow flexion beyond 90° and concomitant vascular injury put these children at increased risk for CS. Mubarak and Carroll5 reported 9 cases of CS in the volar compartment of the forearm after an extension-type supracondylar humeral fracture and attributed 8 of them to elbow flexion beyond 90° after closed reduction. In 29 children with supracondylar humeral fracture,Battaglia and colleagues3 found the highest compartment pressure in the deep volar compartment, especially near the fracture site, as well as a significant increase in pressure with the elbow flexed beyond 90°.
In a study of children with supracondylar humeral fracture, Choi and colleagues6 found 2 cases of CS among 9 patients who presented with a pulseless, poorly perfused hand and no cases of CS among 24 patients who presented with a pulseless but well-perfused hand.
Studies have found that a treatment delay of 8 to 12 hours did not increase the rate of CS in Gartland type 2 and type 3 fractures.7-10 The investigators in these studies did not recommend delaying treatment of patients with neurologic deficit and absent radial pulse. Ramachandran and colleagues4 reported 11 cases of CS in patients with low-energy supracondylar humeral fracture and intact radial pulse at presentation. The patients who developed CS presented with severe swelling, and their mean treatment delay was 22 hours (range, 6-64 hours). Given the data, we do not recommend delayed treatment for children with supracondylar humeral fracture and neurologic deficit or absent pulse. We do recommend close inpatient preoperative monitoring of patients with severe swelling.
CS after supracondylar humeral fracture is mostly seen in the volar compartment of the forearm, but it has also been reported in the mobile wad, the anterior arm compartment, and the posterior arm compartment.11,12
Floating Elbow
CS has been reported in children with ipsilateral humeral and forearm fractures. Blakemore and colleagues13 reported a 33% rate of CS in children with displaced distal humeral and forearm fractures. A retrospective review of 16 cases of floating elbow treated at Boston Children’s Hospital found CS in 2 patients and incipient CS in 4 of 10 patients with forearm fractures treated with closed reduction and plaster casting. There were no signs of CS in 6 patients with distal humeral and forearm fractures stabilized with Kirschner wires.14 Given the data, we do not recommend circumferential casting for forearm fractures in children with floating elbow.
Forearm Fracture
Haasbeek and Cole15 reported CS in 5 (11%) of 46 children with open forearm fracture. Yuan and colleagues16 reported CS in 3 (6%) of 50 open forearm fractures and 3 of 30 closed fractures treated with closed reduction and intramedullary nailing. They found increased risk for CS in patients with longer operative time, indicating prolonged closed manipulation of these fractures as a risk factor for CS. They did not find any cases of CS among 205 forearm fractures treated with closed reduction and casting.
Flynn and colleagues17 reported CS in 2 of 30 patients treated with intramedullary nailing within 24 hours of injury and in 0 of 73 patients treated after 24 hours.
Blackman and colleagues18 reported CS in 3 (7.7%) of 39 open forearm fractures and 0 of 74 closed fractures treated operatively. In their series, a small incision was made to facilitate reduction in 38 (51.4%) of 74 closed fractures to decrease closed manipulation and operative time. The rate of CS after intramedullary nailing of closed forearm fractures was lower in this series than in similar reports in the literature.
Reported data indicate increased risk for CS in children with open forearm fractures and fractures treated with closed reduction and intramedullary nailing, especially performed within 24 hours of injury, and prolonged closed manipulation performed during surgery. We recommend close monitoring of all children with operatively treated forearm fractures and, in particular, children with the risk factors mentioned.
Femoral Fracture
Although CS after femoral shaft fractures is not common, CS has been reported after 90/90 spica casting of femoral shaft fractures in children. Mubarak and colleagues19 reported on 9 children who developed calf CS after treatment of femoral shaft fracture in 90/90 spica casts. The technique used in 7 of the 9 reported cases involved initial application of a short leg cast and then traction applied to the leg—believed to cause impinging of the cast on the posterior compartment of the leg. The authors recommended an alternative method of applying spica casts, which is beyond the scope of this review.
Tibial Fracture
Children with tibial fracture, especially a fracture sustained in a motor vehicle accident, are at risk for CS. Hope and Cole20 found CS in 4 (4%) of 92 children with open tibial fracture.
Children with tibial tubercle fracture are at increased risk for CS because of concomitant vascular injury. Pandya and colleagues21 reported CS or vascular compromise in 4 of 40 patients with tibial tubercle fracture. We recommend close monitoring for signs of impending CS in children who present with high-energy tibial shaft fracture and tibial tubercle fracture.
Flynn and colleagues22 reported outcomes of 43 cases of acute CS of the leg in children treated at 2 pediatric trauma centers. Mean time from injury to fasciotomy was 20.5 hours (range, 3.9-118 hours). Functional outcome was excellent at time of follow-up; 41 of 43 cases had no sequelae, and the 2 patients who lost function underwent fasciotomy more than 80 hours after injury. Despite the long interval between injury and surgery, excellent results were achieved with fasciotomy, suggesting an increased potential for recovery in the pediatric population.
Mubarak23 reported on 6 cases of distal tibial physis fracture in patients who presented with severe pain and swelling of the ankle, hyposthesia of the first web space, weakness of the extensor hallucis longus and extensor digitorum communis, and pain on passive flexion of the toes. In all these patients, intramuscular pressure was more than 40 mm Hg beneath the extensor retinaculum and less than 20 mm Hg in the anterior compartment. All patients experienced prompt relief of pain and improved sensation and strength within 24 hours after release of the superior extensor retinaculum and fracture stabilization.
Miscellaneous and Nontraumatic Causes of Compartment Syndrome
Neonatal CS is very rare, and diagnosis is often missed. Neonatal CS is thought to be caused by a combination of low neonatal blood pressure and birth trauma.24 Ragland and colleagues25 reported on 24 cases of neonatal CS; in only 1 case was the diagnosis made within 24 hours.They described a “sentinel skin lesion” on the forearm of each patient as the sign of neonatal CS. Late diagnosis results in contracture and growth arrest of the involved extremity. In their series, only 1 patient underwent fasciotomy within 24 hours, and it resulted in a good functional outcome. High clinical suspicion is the key to early diagnosis and treatment of this rare pathology.
Medical problems that cause intracompartmental bleeding (hepatic failure, renal failure, leukemia, hemophilia) have been cited as causing CS.26-28 CS may be the first symptom of occult hemophilia29 Correction of the coagulation defect may take priority over surgical treatment in these cases, though the decision should be made on a case-by-case basis.26
CS in children can also be caused by snakebites. Shaw and Hosalkar30 reported on successful use of antivenin in preventing the need for surgical treatment in 16 of 19 patients with rattlesnake bites. Two patients had limited surgical débridement, and 1 underwent fasciotomy for CS. The authors recommended using antivenin to prevent CS in children with snakebites.30
Prasarn and colleagues2 reported on 12 cases of upper extremity CS in children in the absence of fractures. Of the 12 patients, 10 were managed in an intensive care unit and had an obtunded sensorium. Etiology in 7 (58%) of the 12 cases was iatrogenic (intravenous infiltration, retained phlebotomy tourniquet). In this series, 4 amputations were performed on affected extremities.
Diagnosis
Identification of evolving CS in a child is difficult because of the child’s limited ability to communicate and anxiety about being examined by a stranger. Orthopedists are trained to look for the 5 Ps (pain, paresthesia, paralysis, pallor, pulselessness) associated with CS. Examining an anxious, frightened young child is difficult, and documenting the degree of pain is not practical in a child who may not be able or willing to communicate effectively.
In a series of 33 children with CS, Bae and colleagues31 found that the 5 Ps were relatively unreliable in making a timely diagnosis. The authors also found that increased analgesic use was documented a mean of 7.3 hours before a change in vascular status and that it was a more sensitive indicator of CS in children. The resulting recommendation is that children at risk for CS be closely monitored for the 3 As (increasing analgesic requirement, anxiety, agitation).32
Regional anesthesia is used to control postoperative pain in adults and children.33,34 Injudicious use may mask the primary symptom (pain) of CS.32,35-38 Use of regional anesthesia in patients at high risk for CS is highly discouraged.
Although CS is a clinical diagnosis, compartment pressure measurements can be useful in making decisions in certain clinical scenarios. In an obtunded child or in a child with severe mental and communication disability, such a measurement can help confirm or rule out the diagnosis.
Normal compartment pressures are higher in children than in adults. Staudt and colleagues39 compared pressures in 4 lower leg compartments of 20 healthy children and 20 healthy adults. Mean pressure varied from 13.3 mm Hg to 16.6 mm Hg in children and from 5.2 mm Hg to 9.7 mm Hg in adults—indicating higher normal pressure in lower leg compartments in children.
Compartment pressures were reported highest within 5 cm of the fracture site.40 When clinically indicated, they should be measured in that area in an injured extremity. The pressure threshold that requires fasciotomy is debatable. Intracompartmental pressures of 30 to 45 mm Hg, or measurements less than 30 mm Hg of diastolic blood pressure (pressure change = diastolic blood pressure – compartment pressure), have been recommended as cutoffs by some authors.41-44 As resting normal compartment pressures are higher in children, these cutoffs cannot be used as reliably in children as in adults. Direct measurement of intracompartmental pressure is invasive and can be difficult in an agitated, awake child. The potential utility of near-infrared spectroscopy in the diagnosis of increased compartment pressure has been reported.45,46 This method uses differential light absorption properties of oxygenated hemoglobin to measure tissue ischemia—similar to the method used in pulse oximetry. Compared with pulse oximetry, near-infrared spectroscopy can sample deeper tissue (3 cm below skin level). Shuler and colleagues45 reported near-infrared spectroscopy findings for 14 adults with acute CS. Lower tissue oxygenation levels correlated with increased intracompartmental pressures, but the authors could not define a cutoff for which near-infrared spectroscopy measurements would indicate significant tissue ischemia. Use of this method in diagnosing CS in children was described in a case report.46
CS remains a clinical diagnosis. Informing family and staff about the signs and symptoms of this syndrome and closely monitoring analgesic use in these patients are crucial. Compartment pressure measurements can be used when the diagnosis is unclear, particularly in noncommunicative patients, but these values should be interpreted with caution.
Treatment
Once CS is diagnosed, emergent fasciotomy and decompression are indicated. Surgeons planning fasciotomy should be aware of the definitive treatment of the CS etiology. Treatment of clotting deficiency in cases caused by excessive bleeding, fracture fixation, and vascular repair may be indicated during fasciotomy and decompression.
Summary
Increased need for analgesics is often the first sign of CS in children and should be considered the sentinel alarm for ongoing tissue necrosis. CS remains a clinical diagnosis, and compartment pressure should be measured only as a confirmatory test in noncommunicative patients or when the diagnosis is unclear. Children with supracondylar humeral fractures, forearm fractures, tibial fractures, and medical risk factors for coagulopathy are at increased risk and should be monitored closely. When the diagnosis is made promptly and the condition is treated with fasciotomy, good long-term clinical results can be expected.
1. Bhattacharyya T, Vrahas MS. The medical-legal aspects of compartment syndrome. J Bone Joint Surg Am. 2004;86(4):864-868.
2. Prasarn ML, Ouellette EA, Livingstone A, Giuffrida AY. Acute pediatric upper extremity compartment syndrome in the absence of fracture. J Pediatr Orthop. 2009;29(3):263-268.
3. Battaglia TC, Armstrong DG, Schwend RM. Factors affecting forearm compartment pressures in children with supracondylar fractures of the humerus. J Pediatr Orthop. 2002;22(4):431-439.
4. Ramachandran M, Skaggs DL, Crawford HA, et al. Delaying treatment of supracondylar fractures in children: has the pendulum swung too far? J Bone Joint Surg Br. 2008;90(9):1228-1233.
5. Mubarak SJ, Carroll NC. Volkmann’s contracture in children: aetiology and prevention. J Bone Joint Surg Br. 1979;61(3):285-293.
6. Choi PD, Melikian R, Skaggs DL. Risk factors for vascular repair and compartment syndrome in the pulseless supracondylar humerus fracture in children. J Pediatr Orthop. 2010;30(1):50-56.
7. Gupta N, Kay RM, Leitch K, Femino JD, Tolo VT, Skaggs DL. Effect of surgical delay on perioperative complications and need for open reduction in supracondylar humerus fractures in children. J Pediatr Orthop. 2004;24(3):245-248.
8. Iyengar SR, Hoffinger SA, Townsend DR. Early versus delayed reduction and pinning of type III displaced supracondylar fractures of the humerus in children: a comparative study. J Orthop Trauma. 1999;13(1):51-55.
9. Leet AI, Frisancho J, Ebramzadeh E. Delayed treatment of type 3 supracondylar humerus fractures in children. J Pediatr Orthop. 2002;22(2):203-207.
10. Mehlman CT, Strub WM, Roy DR, Wall EJ, Crawford AH. The effect of surgical timing on the perioperative complications of treatment of supracondylar humeral fractures in children. J Bone Joint Surg Am. 2001;83(3):323-327.
11. Diesselhorst MM, Deck JW, Davey JP. Compartment syndrome of the upper arm after closed reduction and percutaneous pinning of a supracondylar humerus fracture. J Pediatr Orthop. 2014;34(2):e1-e4.
12. Mai MC, Beck R, Gabriel K, Singh KA. Posterior arm compartment syndrome after a combined supracondylar humeral and capitellar fractures in an adolescent: a case report. J Pediatr Orthop. 2011;31(3):e16-e19.
13. Blakemore LC, Cooperman DR, Thompson GH, Wathey C, Ballock RT. Compartment syndrome in ipsilateral humerus and forearm fractures in children. Clin Orthop Relat Res. 2000;(376):32-38.
14. Ring D, Waters PM, Hotchkiss RN, Kasser JR. Pediatric floating elbow. J Pediatr Orthop. 2001;21(4):456-459.
15. Haasbeek JF, Cole WG. Open fractures of the arm in children. J Bone Joint Surg Br. 1995;77(4):576-581.
16. Yuan PS, Pring ME, Gaynor TP, Mubarak SJ, Newton PO. Compartment syndrome following intramedullary fixation of pediatric forearm fractures. J Pediatr Orthop. 2004;24(4):370-375.
17. Flynn JM, Jones KJ, Garner MR, Goebel J. Eleven years experience in the operative management of pediatric forearm fractures. J Pediatr Orthop. 2010;30(4):313-319.
18. Blackman AJ, Wall LB, Keeler KA, et al. Acute compartment syndrome after intramedullary nailing of isolated radius and ulna fractures in children. J Pediatr Orthop. 2014;34(1):50-54.
19. Mubarak SJ, Frick S, Sink E, Rathjen K, Noonan KJ. Volkmann contracture and compartment syndromes after femur fractures in children treated with 90/90 spica casts. J Pediatr Orthop. 2006;26(5):567-572.
20. Hope PG, Cole WG. Open fractures of the tibia in children. J Bone Joint Surg Br. 1992;74(4):546-553.
21. Pandya NK, Edmonds EK, Roocroft JH, Mubarak SJ. Tibial tubercle fractures: complications, classification, and the need for intra-articular assessment. J Pediatr Orthop. 2012;32(8):749-759.
22. Flynn JM, Bashyal RK, Yeger-McKeever M, Garner MR, Launay F, Sponseller PD. Acute traumatic compartment syndrome of the leg in children: diagnosis and outcome. J Bone Joint Surg Am. 2011;93(10):937-941.
23. Mubarak SJ. Extensor retinaculum syndrome of the ankle after injury to the distal tibial physis. J Bone Joint Surg Br. 2002;84(1):11-14.
24. Macer GA Jr. Forearm compartment syndrome in the newborn. J Hand Surg Am. 2006;31(9):1550.
25. Ragland R 3rd, Moukoko D, Ezaki M, Carter PR, Mills J. Forearm compartment syndrome in the newborn: report of 24 cases. J Hand Surg Am. 2005;30(5):997-1003.
26. Alioglu B, Avci Z, Baskin E, Ozcay F, Tuncay IC, Ozbek N. Successful use of recombinant factor VIIa (NovoSeven) in children with compartment syndrome: two case reports. J Pediatr Orthop. 2006;26(6):815-817.
27. Lee DK, Jeong WK, Lee DH, Lee SH. Multiple compartment syndrome in a pediatric patient with CML. J Pediatr Orthop. 2011;31(8):889-892.
28. Dumontier C, Sautet A, Man M, Bennani M, Apoil A. Entrapment and compartment syndromes of the upper limb in haemophilia. J Hand Surg Br. 1994;19(4):427-429.
29. Jones G, Thompson K, Johnson M. Acute compartment syndrome after minor trauma in a patient with undiagnosed mild haemophilia B. Lancet. 2013;382(9905):1678.
30. Shaw BA, Hosalkar HS. Rattlesnake bites in children: antivenin treatment and surgical indications. J Bone Joint Surg Am. 2002;84(9):1624-1629.
31. Bae DS, Kadiyala RK, Waters PM. Acute compartment syndrome in children: contemporary diagnosis, treatment, and outcome. J Pediatr Orthop. 2001;21(5):680-688.
32. Noonan KJ, McCarthy JJ. Compartment syndromes in the pediatric patient. J Pediatr Orthop. 2010;30(2 suppl):S96-S101.
33. Dalens B. Some current controversies in paediatric regional anaesthesia. Curr Opin Anaesthesiol. 2006;19(3):301-308.
34. Wedel DJ. Regional anesthesia and pain management: reviewing the past decade and predicting the future. Anesth Analg. 2000;90(5):1244-1245.
35. Mubarak SJ. Wilton NC. Compartment syndromes and epidural analgesia. J Pediatr Orthop. 1997;17(3):282-284.
36. Price C, Ribeiro J, Kinnebrew T. Compartment syndromes associated with postoperative epidural analgesia. A case report. J Bone Joint Surg Am. 1996;78(4):597-599.
37. Thonse R, Ashford RU, Williams TI, Harrington P. Differences in attitudes to analgesia in post-operative limb surgery put patients at risk of compartment syndrome. Injury. 2004;35(3):290-295.
38. Whitesides TE Jr. Pain: friend or foe? J Bone Joint Surg Am. 2001;83(9):1424-1425.
39. Staudt JM, Smeulders MJ, van der Horst CM. Normal compartment pressures of the lower leg in children. J Bone Joint Surg Br. 2008;90(2):215-219.
40. Heckman MM, Whitesides TE Jr, Grewe SR, Rooks MD. Compartment pressure in association with closed tibial fractures. The relationship between tissue pressure, compartment, and the distance from the site of the fracture. J Bone Joint Surg Am. 1994;76(9):1285-1292.
41. Hargens AR, Schmidt DA, Evans KL, et al. Quantitation of skeletal-muscle necrosis in a model compartment syndrome. J Bone Joint Surg Am. 1981;63(4):631-636.
42. Heppenstall RB, Sapega AA, Scott R, et al. The compartment syndrome. An experimental and clinical study of muscular energy metabolism using phosphorus nuclear magnetic resonance spectroscopy. Clin Orthop Relat Res. 1988;(226):138-155.
43. McQueen MM, Court-Brown CM. Compartment monitoring in tibial fractures. The pressure threshold for decompression. J Bone Joint Surg Br. 1996;78(1):99-104.
44. Rorabeck CH. The treatment of compartment syndromes of the leg. J Bone Joint Surg Br. 1984;66(1):93-97.
45. Shuler MS, Reisman WM, Kinsey TL, et al. Correlation between muscle oxygenation and compartment pressures in acute compartment syndrome of the leg. J Bone Joint Surg Am. 2010;92(4):863-870.
46. Tobias JD, Hoernschemeyer DG. Near-infrared spectroscopy identifies compartment syndrome in an infant. J Pediatr Orthop. 2007;27(3):311-313.
Compartment syndrome (CS) is one of the true orthopedic emergencies. Identifying the high-risk patient, making a prompt diagnosis, and initiating effective treatment are the crucial steps in avoiding a poor outcome. A physician’s inability to communicate with young children can interfere with diagnosing CS in a timely fashion. Many young patients in hospitals are admitted to pediatric floors where routine orthopedic care is not the norm and staff are unfamiliar with the signs and symptoms of evolving CS. As orthopedic surgeons are often involved in caring for these patients, they should be aware of the aspects of CS that are unique to children and should be able to identify patients who are at risk and would benefit from close monitoring. In addition, given the consequences of late diagnosis, early diagnosis is important from a medicolegal standpoint. Only 44% of cases of adult and pediatric CS are decided in favor of treating physicians, compared with 75% of cases in other orthopedic malpractice claims.1,2
Risk Factors for Posttraumatic Compartment Syndrome
Supracondylar Humeral Fracture
CS is a well-described complication of this injury. CS develops in 0.1% to 0.3% of children who present with supracondylar humeral fracture.3,4 Casted elbow flexion beyond 90° and concomitant vascular injury put these children at increased risk for CS. Mubarak and Carroll5 reported 9 cases of CS in the volar compartment of the forearm after an extension-type supracondylar humeral fracture and attributed 8 of them to elbow flexion beyond 90° after closed reduction. In 29 children with supracondylar humeral fracture,Battaglia and colleagues3 found the highest compartment pressure in the deep volar compartment, especially near the fracture site, as well as a significant increase in pressure with the elbow flexed beyond 90°.
In a study of children with supracondylar humeral fracture, Choi and colleagues6 found 2 cases of CS among 9 patients who presented with a pulseless, poorly perfused hand and no cases of CS among 24 patients who presented with a pulseless but well-perfused hand.
Studies have found that a treatment delay of 8 to 12 hours did not increase the rate of CS in Gartland type 2 and type 3 fractures.7-10 The investigators in these studies did not recommend delaying treatment of patients with neurologic deficit and absent radial pulse. Ramachandran and colleagues4 reported 11 cases of CS in patients with low-energy supracondylar humeral fracture and intact radial pulse at presentation. The patients who developed CS presented with severe swelling, and their mean treatment delay was 22 hours (range, 6-64 hours). Given the data, we do not recommend delayed treatment for children with supracondylar humeral fracture and neurologic deficit or absent pulse. We do recommend close inpatient preoperative monitoring of patients with severe swelling.
CS after supracondylar humeral fracture is mostly seen in the volar compartment of the forearm, but it has also been reported in the mobile wad, the anterior arm compartment, and the posterior arm compartment.11,12
Floating Elbow
CS has been reported in children with ipsilateral humeral and forearm fractures. Blakemore and colleagues13 reported a 33% rate of CS in children with displaced distal humeral and forearm fractures. A retrospective review of 16 cases of floating elbow treated at Boston Children’s Hospital found CS in 2 patients and incipient CS in 4 of 10 patients with forearm fractures treated with closed reduction and plaster casting. There were no signs of CS in 6 patients with distal humeral and forearm fractures stabilized with Kirschner wires.14 Given the data, we do not recommend circumferential casting for forearm fractures in children with floating elbow.
Forearm Fracture
Haasbeek and Cole15 reported CS in 5 (11%) of 46 children with open forearm fracture. Yuan and colleagues16 reported CS in 3 (6%) of 50 open forearm fractures and 3 of 30 closed fractures treated with closed reduction and intramedullary nailing. They found increased risk for CS in patients with longer operative time, indicating prolonged closed manipulation of these fractures as a risk factor for CS. They did not find any cases of CS among 205 forearm fractures treated with closed reduction and casting.
Flynn and colleagues17 reported CS in 2 of 30 patients treated with intramedullary nailing within 24 hours of injury and in 0 of 73 patients treated after 24 hours.
Blackman and colleagues18 reported CS in 3 (7.7%) of 39 open forearm fractures and 0 of 74 closed fractures treated operatively. In their series, a small incision was made to facilitate reduction in 38 (51.4%) of 74 closed fractures to decrease closed manipulation and operative time. The rate of CS after intramedullary nailing of closed forearm fractures was lower in this series than in similar reports in the literature.
Reported data indicate increased risk for CS in children with open forearm fractures and fractures treated with closed reduction and intramedullary nailing, especially performed within 24 hours of injury, and prolonged closed manipulation performed during surgery. We recommend close monitoring of all children with operatively treated forearm fractures and, in particular, children with the risk factors mentioned.
Femoral Fracture
Although CS after femoral shaft fractures is not common, CS has been reported after 90/90 spica casting of femoral shaft fractures in children. Mubarak and colleagues19 reported on 9 children who developed calf CS after treatment of femoral shaft fracture in 90/90 spica casts. The technique used in 7 of the 9 reported cases involved initial application of a short leg cast and then traction applied to the leg—believed to cause impinging of the cast on the posterior compartment of the leg. The authors recommended an alternative method of applying spica casts, which is beyond the scope of this review.
Tibial Fracture
Children with tibial fracture, especially a fracture sustained in a motor vehicle accident, are at risk for CS. Hope and Cole20 found CS in 4 (4%) of 92 children with open tibial fracture.
Children with tibial tubercle fracture are at increased risk for CS because of concomitant vascular injury. Pandya and colleagues21 reported CS or vascular compromise in 4 of 40 patients with tibial tubercle fracture. We recommend close monitoring for signs of impending CS in children who present with high-energy tibial shaft fracture and tibial tubercle fracture.
Flynn and colleagues22 reported outcomes of 43 cases of acute CS of the leg in children treated at 2 pediatric trauma centers. Mean time from injury to fasciotomy was 20.5 hours (range, 3.9-118 hours). Functional outcome was excellent at time of follow-up; 41 of 43 cases had no sequelae, and the 2 patients who lost function underwent fasciotomy more than 80 hours after injury. Despite the long interval between injury and surgery, excellent results were achieved with fasciotomy, suggesting an increased potential for recovery in the pediatric population.
Mubarak23 reported on 6 cases of distal tibial physis fracture in patients who presented with severe pain and swelling of the ankle, hyposthesia of the first web space, weakness of the extensor hallucis longus and extensor digitorum communis, and pain on passive flexion of the toes. In all these patients, intramuscular pressure was more than 40 mm Hg beneath the extensor retinaculum and less than 20 mm Hg in the anterior compartment. All patients experienced prompt relief of pain and improved sensation and strength within 24 hours after release of the superior extensor retinaculum and fracture stabilization.
Miscellaneous and Nontraumatic Causes of Compartment Syndrome
Neonatal CS is very rare, and diagnosis is often missed. Neonatal CS is thought to be caused by a combination of low neonatal blood pressure and birth trauma.24 Ragland and colleagues25 reported on 24 cases of neonatal CS; in only 1 case was the diagnosis made within 24 hours.They described a “sentinel skin lesion” on the forearm of each patient as the sign of neonatal CS. Late diagnosis results in contracture and growth arrest of the involved extremity. In their series, only 1 patient underwent fasciotomy within 24 hours, and it resulted in a good functional outcome. High clinical suspicion is the key to early diagnosis and treatment of this rare pathology.
Medical problems that cause intracompartmental bleeding (hepatic failure, renal failure, leukemia, hemophilia) have been cited as causing CS.26-28 CS may be the first symptom of occult hemophilia29 Correction of the coagulation defect may take priority over surgical treatment in these cases, though the decision should be made on a case-by-case basis.26
CS in children can also be caused by snakebites. Shaw and Hosalkar30 reported on successful use of antivenin in preventing the need for surgical treatment in 16 of 19 patients with rattlesnake bites. Two patients had limited surgical débridement, and 1 underwent fasciotomy for CS. The authors recommended using antivenin to prevent CS in children with snakebites.30
Prasarn and colleagues2 reported on 12 cases of upper extremity CS in children in the absence of fractures. Of the 12 patients, 10 were managed in an intensive care unit and had an obtunded sensorium. Etiology in 7 (58%) of the 12 cases was iatrogenic (intravenous infiltration, retained phlebotomy tourniquet). In this series, 4 amputations were performed on affected extremities.
Diagnosis
Identification of evolving CS in a child is difficult because of the child’s limited ability to communicate and anxiety about being examined by a stranger. Orthopedists are trained to look for the 5 Ps (pain, paresthesia, paralysis, pallor, pulselessness) associated with CS. Examining an anxious, frightened young child is difficult, and documenting the degree of pain is not practical in a child who may not be able or willing to communicate effectively.
In a series of 33 children with CS, Bae and colleagues31 found that the 5 Ps were relatively unreliable in making a timely diagnosis. The authors also found that increased analgesic use was documented a mean of 7.3 hours before a change in vascular status and that it was a more sensitive indicator of CS in children. The resulting recommendation is that children at risk for CS be closely monitored for the 3 As (increasing analgesic requirement, anxiety, agitation).32
Regional anesthesia is used to control postoperative pain in adults and children.33,34 Injudicious use may mask the primary symptom (pain) of CS.32,35-38 Use of regional anesthesia in patients at high risk for CS is highly discouraged.
Although CS is a clinical diagnosis, compartment pressure measurements can be useful in making decisions in certain clinical scenarios. In an obtunded child or in a child with severe mental and communication disability, such a measurement can help confirm or rule out the diagnosis.
Normal compartment pressures are higher in children than in adults. Staudt and colleagues39 compared pressures in 4 lower leg compartments of 20 healthy children and 20 healthy adults. Mean pressure varied from 13.3 mm Hg to 16.6 mm Hg in children and from 5.2 mm Hg to 9.7 mm Hg in adults—indicating higher normal pressure in lower leg compartments in children.
Compartment pressures were reported highest within 5 cm of the fracture site.40 When clinically indicated, they should be measured in that area in an injured extremity. The pressure threshold that requires fasciotomy is debatable. Intracompartmental pressures of 30 to 45 mm Hg, or measurements less than 30 mm Hg of diastolic blood pressure (pressure change = diastolic blood pressure – compartment pressure), have been recommended as cutoffs by some authors.41-44 As resting normal compartment pressures are higher in children, these cutoffs cannot be used as reliably in children as in adults. Direct measurement of intracompartmental pressure is invasive and can be difficult in an agitated, awake child. The potential utility of near-infrared spectroscopy in the diagnosis of increased compartment pressure has been reported.45,46 This method uses differential light absorption properties of oxygenated hemoglobin to measure tissue ischemia—similar to the method used in pulse oximetry. Compared with pulse oximetry, near-infrared spectroscopy can sample deeper tissue (3 cm below skin level). Shuler and colleagues45 reported near-infrared spectroscopy findings for 14 adults with acute CS. Lower tissue oxygenation levels correlated with increased intracompartmental pressures, but the authors could not define a cutoff for which near-infrared spectroscopy measurements would indicate significant tissue ischemia. Use of this method in diagnosing CS in children was described in a case report.46
CS remains a clinical diagnosis. Informing family and staff about the signs and symptoms of this syndrome and closely monitoring analgesic use in these patients are crucial. Compartment pressure measurements can be used when the diagnosis is unclear, particularly in noncommunicative patients, but these values should be interpreted with caution.
Treatment
Once CS is diagnosed, emergent fasciotomy and decompression are indicated. Surgeons planning fasciotomy should be aware of the definitive treatment of the CS etiology. Treatment of clotting deficiency in cases caused by excessive bleeding, fracture fixation, and vascular repair may be indicated during fasciotomy and decompression.
Summary
Increased need for analgesics is often the first sign of CS in children and should be considered the sentinel alarm for ongoing tissue necrosis. CS remains a clinical diagnosis, and compartment pressure should be measured only as a confirmatory test in noncommunicative patients or when the diagnosis is unclear. Children with supracondylar humeral fractures, forearm fractures, tibial fractures, and medical risk factors for coagulopathy are at increased risk and should be monitored closely. When the diagnosis is made promptly and the condition is treated with fasciotomy, good long-term clinical results can be expected.
Compartment syndrome (CS) is one of the true orthopedic emergencies. Identifying the high-risk patient, making a prompt diagnosis, and initiating effective treatment are the crucial steps in avoiding a poor outcome. A physician’s inability to communicate with young children can interfere with diagnosing CS in a timely fashion. Many young patients in hospitals are admitted to pediatric floors where routine orthopedic care is not the norm and staff are unfamiliar with the signs and symptoms of evolving CS. As orthopedic surgeons are often involved in caring for these patients, they should be aware of the aspects of CS that are unique to children and should be able to identify patients who are at risk and would benefit from close monitoring. In addition, given the consequences of late diagnosis, early diagnosis is important from a medicolegal standpoint. Only 44% of cases of adult and pediatric CS are decided in favor of treating physicians, compared with 75% of cases in other orthopedic malpractice claims.1,2
Risk Factors for Posttraumatic Compartment Syndrome
Supracondylar Humeral Fracture
CS is a well-described complication of this injury. CS develops in 0.1% to 0.3% of children who present with supracondylar humeral fracture.3,4 Casted elbow flexion beyond 90° and concomitant vascular injury put these children at increased risk for CS. Mubarak and Carroll5 reported 9 cases of CS in the volar compartment of the forearm after an extension-type supracondylar humeral fracture and attributed 8 of them to elbow flexion beyond 90° after closed reduction. In 29 children with supracondylar humeral fracture,Battaglia and colleagues3 found the highest compartment pressure in the deep volar compartment, especially near the fracture site, as well as a significant increase in pressure with the elbow flexed beyond 90°.
In a study of children with supracondylar humeral fracture, Choi and colleagues6 found 2 cases of CS among 9 patients who presented with a pulseless, poorly perfused hand and no cases of CS among 24 patients who presented with a pulseless but well-perfused hand.
Studies have found that a treatment delay of 8 to 12 hours did not increase the rate of CS in Gartland type 2 and type 3 fractures.7-10 The investigators in these studies did not recommend delaying treatment of patients with neurologic deficit and absent radial pulse. Ramachandran and colleagues4 reported 11 cases of CS in patients with low-energy supracondylar humeral fracture and intact radial pulse at presentation. The patients who developed CS presented with severe swelling, and their mean treatment delay was 22 hours (range, 6-64 hours). Given the data, we do not recommend delayed treatment for children with supracondylar humeral fracture and neurologic deficit or absent pulse. We do recommend close inpatient preoperative monitoring of patients with severe swelling.
CS after supracondylar humeral fracture is mostly seen in the volar compartment of the forearm, but it has also been reported in the mobile wad, the anterior arm compartment, and the posterior arm compartment.11,12
Floating Elbow
CS has been reported in children with ipsilateral humeral and forearm fractures. Blakemore and colleagues13 reported a 33% rate of CS in children with displaced distal humeral and forearm fractures. A retrospective review of 16 cases of floating elbow treated at Boston Children’s Hospital found CS in 2 patients and incipient CS in 4 of 10 patients with forearm fractures treated with closed reduction and plaster casting. There were no signs of CS in 6 patients with distal humeral and forearm fractures stabilized with Kirschner wires.14 Given the data, we do not recommend circumferential casting for forearm fractures in children with floating elbow.
Forearm Fracture
Haasbeek and Cole15 reported CS in 5 (11%) of 46 children with open forearm fracture. Yuan and colleagues16 reported CS in 3 (6%) of 50 open forearm fractures and 3 of 30 closed fractures treated with closed reduction and intramedullary nailing. They found increased risk for CS in patients with longer operative time, indicating prolonged closed manipulation of these fractures as a risk factor for CS. They did not find any cases of CS among 205 forearm fractures treated with closed reduction and casting.
Flynn and colleagues17 reported CS in 2 of 30 patients treated with intramedullary nailing within 24 hours of injury and in 0 of 73 patients treated after 24 hours.
Blackman and colleagues18 reported CS in 3 (7.7%) of 39 open forearm fractures and 0 of 74 closed fractures treated operatively. In their series, a small incision was made to facilitate reduction in 38 (51.4%) of 74 closed fractures to decrease closed manipulation and operative time. The rate of CS after intramedullary nailing of closed forearm fractures was lower in this series than in similar reports in the literature.
Reported data indicate increased risk for CS in children with open forearm fractures and fractures treated with closed reduction and intramedullary nailing, especially performed within 24 hours of injury, and prolonged closed manipulation performed during surgery. We recommend close monitoring of all children with operatively treated forearm fractures and, in particular, children with the risk factors mentioned.
Femoral Fracture
Although CS after femoral shaft fractures is not common, CS has been reported after 90/90 spica casting of femoral shaft fractures in children. Mubarak and colleagues19 reported on 9 children who developed calf CS after treatment of femoral shaft fracture in 90/90 spica casts. The technique used in 7 of the 9 reported cases involved initial application of a short leg cast and then traction applied to the leg—believed to cause impinging of the cast on the posterior compartment of the leg. The authors recommended an alternative method of applying spica casts, which is beyond the scope of this review.
Tibial Fracture
Children with tibial fracture, especially a fracture sustained in a motor vehicle accident, are at risk for CS. Hope and Cole20 found CS in 4 (4%) of 92 children with open tibial fracture.
Children with tibial tubercle fracture are at increased risk for CS because of concomitant vascular injury. Pandya and colleagues21 reported CS or vascular compromise in 4 of 40 patients with tibial tubercle fracture. We recommend close monitoring for signs of impending CS in children who present with high-energy tibial shaft fracture and tibial tubercle fracture.
Flynn and colleagues22 reported outcomes of 43 cases of acute CS of the leg in children treated at 2 pediatric trauma centers. Mean time from injury to fasciotomy was 20.5 hours (range, 3.9-118 hours). Functional outcome was excellent at time of follow-up; 41 of 43 cases had no sequelae, and the 2 patients who lost function underwent fasciotomy more than 80 hours after injury. Despite the long interval between injury and surgery, excellent results were achieved with fasciotomy, suggesting an increased potential for recovery in the pediatric population.
Mubarak23 reported on 6 cases of distal tibial physis fracture in patients who presented with severe pain and swelling of the ankle, hyposthesia of the first web space, weakness of the extensor hallucis longus and extensor digitorum communis, and pain on passive flexion of the toes. In all these patients, intramuscular pressure was more than 40 mm Hg beneath the extensor retinaculum and less than 20 mm Hg in the anterior compartment. All patients experienced prompt relief of pain and improved sensation and strength within 24 hours after release of the superior extensor retinaculum and fracture stabilization.
Miscellaneous and Nontraumatic Causes of Compartment Syndrome
Neonatal CS is very rare, and diagnosis is often missed. Neonatal CS is thought to be caused by a combination of low neonatal blood pressure and birth trauma.24 Ragland and colleagues25 reported on 24 cases of neonatal CS; in only 1 case was the diagnosis made within 24 hours.They described a “sentinel skin lesion” on the forearm of each patient as the sign of neonatal CS. Late diagnosis results in contracture and growth arrest of the involved extremity. In their series, only 1 patient underwent fasciotomy within 24 hours, and it resulted in a good functional outcome. High clinical suspicion is the key to early diagnosis and treatment of this rare pathology.
Medical problems that cause intracompartmental bleeding (hepatic failure, renal failure, leukemia, hemophilia) have been cited as causing CS.26-28 CS may be the first symptom of occult hemophilia29 Correction of the coagulation defect may take priority over surgical treatment in these cases, though the decision should be made on a case-by-case basis.26
CS in children can also be caused by snakebites. Shaw and Hosalkar30 reported on successful use of antivenin in preventing the need for surgical treatment in 16 of 19 patients with rattlesnake bites. Two patients had limited surgical débridement, and 1 underwent fasciotomy for CS. The authors recommended using antivenin to prevent CS in children with snakebites.30
Prasarn and colleagues2 reported on 12 cases of upper extremity CS in children in the absence of fractures. Of the 12 patients, 10 were managed in an intensive care unit and had an obtunded sensorium. Etiology in 7 (58%) of the 12 cases was iatrogenic (intravenous infiltration, retained phlebotomy tourniquet). In this series, 4 amputations were performed on affected extremities.
Diagnosis
Identification of evolving CS in a child is difficult because of the child’s limited ability to communicate and anxiety about being examined by a stranger. Orthopedists are trained to look for the 5 Ps (pain, paresthesia, paralysis, pallor, pulselessness) associated with CS. Examining an anxious, frightened young child is difficult, and documenting the degree of pain is not practical in a child who may not be able or willing to communicate effectively.
In a series of 33 children with CS, Bae and colleagues31 found that the 5 Ps were relatively unreliable in making a timely diagnosis. The authors also found that increased analgesic use was documented a mean of 7.3 hours before a change in vascular status and that it was a more sensitive indicator of CS in children. The resulting recommendation is that children at risk for CS be closely monitored for the 3 As (increasing analgesic requirement, anxiety, agitation).32
Regional anesthesia is used to control postoperative pain in adults and children.33,34 Injudicious use may mask the primary symptom (pain) of CS.32,35-38 Use of regional anesthesia in patients at high risk for CS is highly discouraged.
Although CS is a clinical diagnosis, compartment pressure measurements can be useful in making decisions in certain clinical scenarios. In an obtunded child or in a child with severe mental and communication disability, such a measurement can help confirm or rule out the diagnosis.
Normal compartment pressures are higher in children than in adults. Staudt and colleagues39 compared pressures in 4 lower leg compartments of 20 healthy children and 20 healthy adults. Mean pressure varied from 13.3 mm Hg to 16.6 mm Hg in children and from 5.2 mm Hg to 9.7 mm Hg in adults—indicating higher normal pressure in lower leg compartments in children.
Compartment pressures were reported highest within 5 cm of the fracture site.40 When clinically indicated, they should be measured in that area in an injured extremity. The pressure threshold that requires fasciotomy is debatable. Intracompartmental pressures of 30 to 45 mm Hg, or measurements less than 30 mm Hg of diastolic blood pressure (pressure change = diastolic blood pressure – compartment pressure), have been recommended as cutoffs by some authors.41-44 As resting normal compartment pressures are higher in children, these cutoffs cannot be used as reliably in children as in adults. Direct measurement of intracompartmental pressure is invasive and can be difficult in an agitated, awake child. The potential utility of near-infrared spectroscopy in the diagnosis of increased compartment pressure has been reported.45,46 This method uses differential light absorption properties of oxygenated hemoglobin to measure tissue ischemia—similar to the method used in pulse oximetry. Compared with pulse oximetry, near-infrared spectroscopy can sample deeper tissue (3 cm below skin level). Shuler and colleagues45 reported near-infrared spectroscopy findings for 14 adults with acute CS. Lower tissue oxygenation levels correlated with increased intracompartmental pressures, but the authors could not define a cutoff for which near-infrared spectroscopy measurements would indicate significant tissue ischemia. Use of this method in diagnosing CS in children was described in a case report.46
CS remains a clinical diagnosis. Informing family and staff about the signs and symptoms of this syndrome and closely monitoring analgesic use in these patients are crucial. Compartment pressure measurements can be used when the diagnosis is unclear, particularly in noncommunicative patients, but these values should be interpreted with caution.
Treatment
Once CS is diagnosed, emergent fasciotomy and decompression are indicated. Surgeons planning fasciotomy should be aware of the definitive treatment of the CS etiology. Treatment of clotting deficiency in cases caused by excessive bleeding, fracture fixation, and vascular repair may be indicated during fasciotomy and decompression.
Summary
Increased need for analgesics is often the first sign of CS in children and should be considered the sentinel alarm for ongoing tissue necrosis. CS remains a clinical diagnosis, and compartment pressure should be measured only as a confirmatory test in noncommunicative patients or when the diagnosis is unclear. Children with supracondylar humeral fractures, forearm fractures, tibial fractures, and medical risk factors for coagulopathy are at increased risk and should be monitored closely. When the diagnosis is made promptly and the condition is treated with fasciotomy, good long-term clinical results can be expected.
1. Bhattacharyya T, Vrahas MS. The medical-legal aspects of compartment syndrome. J Bone Joint Surg Am. 2004;86(4):864-868.
2. Prasarn ML, Ouellette EA, Livingstone A, Giuffrida AY. Acute pediatric upper extremity compartment syndrome in the absence of fracture. J Pediatr Orthop. 2009;29(3):263-268.
3. Battaglia TC, Armstrong DG, Schwend RM. Factors affecting forearm compartment pressures in children with supracondylar fractures of the humerus. J Pediatr Orthop. 2002;22(4):431-439.
4. Ramachandran M, Skaggs DL, Crawford HA, et al. Delaying treatment of supracondylar fractures in children: has the pendulum swung too far? J Bone Joint Surg Br. 2008;90(9):1228-1233.
5. Mubarak SJ, Carroll NC. Volkmann’s contracture in children: aetiology and prevention. J Bone Joint Surg Br. 1979;61(3):285-293.
6. Choi PD, Melikian R, Skaggs DL. Risk factors for vascular repair and compartment syndrome in the pulseless supracondylar humerus fracture in children. J Pediatr Orthop. 2010;30(1):50-56.
7. Gupta N, Kay RM, Leitch K, Femino JD, Tolo VT, Skaggs DL. Effect of surgical delay on perioperative complications and need for open reduction in supracondylar humerus fractures in children. J Pediatr Orthop. 2004;24(3):245-248.
8. Iyengar SR, Hoffinger SA, Townsend DR. Early versus delayed reduction and pinning of type III displaced supracondylar fractures of the humerus in children: a comparative study. J Orthop Trauma. 1999;13(1):51-55.
9. Leet AI, Frisancho J, Ebramzadeh E. Delayed treatment of type 3 supracondylar humerus fractures in children. J Pediatr Orthop. 2002;22(2):203-207.
10. Mehlman CT, Strub WM, Roy DR, Wall EJ, Crawford AH. The effect of surgical timing on the perioperative complications of treatment of supracondylar humeral fractures in children. J Bone Joint Surg Am. 2001;83(3):323-327.
11. Diesselhorst MM, Deck JW, Davey JP. Compartment syndrome of the upper arm after closed reduction and percutaneous pinning of a supracondylar humerus fracture. J Pediatr Orthop. 2014;34(2):e1-e4.
12. Mai MC, Beck R, Gabriel K, Singh KA. Posterior arm compartment syndrome after a combined supracondylar humeral and capitellar fractures in an adolescent: a case report. J Pediatr Orthop. 2011;31(3):e16-e19.
13. Blakemore LC, Cooperman DR, Thompson GH, Wathey C, Ballock RT. Compartment syndrome in ipsilateral humerus and forearm fractures in children. Clin Orthop Relat Res. 2000;(376):32-38.
14. Ring D, Waters PM, Hotchkiss RN, Kasser JR. Pediatric floating elbow. J Pediatr Orthop. 2001;21(4):456-459.
15. Haasbeek JF, Cole WG. Open fractures of the arm in children. J Bone Joint Surg Br. 1995;77(4):576-581.
16. Yuan PS, Pring ME, Gaynor TP, Mubarak SJ, Newton PO. Compartment syndrome following intramedullary fixation of pediatric forearm fractures. J Pediatr Orthop. 2004;24(4):370-375.
17. Flynn JM, Jones KJ, Garner MR, Goebel J. Eleven years experience in the operative management of pediatric forearm fractures. J Pediatr Orthop. 2010;30(4):313-319.
18. Blackman AJ, Wall LB, Keeler KA, et al. Acute compartment syndrome after intramedullary nailing of isolated radius and ulna fractures in children. J Pediatr Orthop. 2014;34(1):50-54.
19. Mubarak SJ, Frick S, Sink E, Rathjen K, Noonan KJ. Volkmann contracture and compartment syndromes after femur fractures in children treated with 90/90 spica casts. J Pediatr Orthop. 2006;26(5):567-572.
20. Hope PG, Cole WG. Open fractures of the tibia in children. J Bone Joint Surg Br. 1992;74(4):546-553.
21. Pandya NK, Edmonds EK, Roocroft JH, Mubarak SJ. Tibial tubercle fractures: complications, classification, and the need for intra-articular assessment. J Pediatr Orthop. 2012;32(8):749-759.
22. Flynn JM, Bashyal RK, Yeger-McKeever M, Garner MR, Launay F, Sponseller PD. Acute traumatic compartment syndrome of the leg in children: diagnosis and outcome. J Bone Joint Surg Am. 2011;93(10):937-941.
23. Mubarak SJ. Extensor retinaculum syndrome of the ankle after injury to the distal tibial physis. J Bone Joint Surg Br. 2002;84(1):11-14.
24. Macer GA Jr. Forearm compartment syndrome in the newborn. J Hand Surg Am. 2006;31(9):1550.
25. Ragland R 3rd, Moukoko D, Ezaki M, Carter PR, Mills J. Forearm compartment syndrome in the newborn: report of 24 cases. J Hand Surg Am. 2005;30(5):997-1003.
26. Alioglu B, Avci Z, Baskin E, Ozcay F, Tuncay IC, Ozbek N. Successful use of recombinant factor VIIa (NovoSeven) in children with compartment syndrome: two case reports. J Pediatr Orthop. 2006;26(6):815-817.
27. Lee DK, Jeong WK, Lee DH, Lee SH. Multiple compartment syndrome in a pediatric patient with CML. J Pediatr Orthop. 2011;31(8):889-892.
28. Dumontier C, Sautet A, Man M, Bennani M, Apoil A. Entrapment and compartment syndromes of the upper limb in haemophilia. J Hand Surg Br. 1994;19(4):427-429.
29. Jones G, Thompson K, Johnson M. Acute compartment syndrome after minor trauma in a patient with undiagnosed mild haemophilia B. Lancet. 2013;382(9905):1678.
30. Shaw BA, Hosalkar HS. Rattlesnake bites in children: antivenin treatment and surgical indications. J Bone Joint Surg Am. 2002;84(9):1624-1629.
31. Bae DS, Kadiyala RK, Waters PM. Acute compartment syndrome in children: contemporary diagnosis, treatment, and outcome. J Pediatr Orthop. 2001;21(5):680-688.
32. Noonan KJ, McCarthy JJ. Compartment syndromes in the pediatric patient. J Pediatr Orthop. 2010;30(2 suppl):S96-S101.
33. Dalens B. Some current controversies in paediatric regional anaesthesia. Curr Opin Anaesthesiol. 2006;19(3):301-308.
34. Wedel DJ. Regional anesthesia and pain management: reviewing the past decade and predicting the future. Anesth Analg. 2000;90(5):1244-1245.
35. Mubarak SJ. Wilton NC. Compartment syndromes and epidural analgesia. J Pediatr Orthop. 1997;17(3):282-284.
36. Price C, Ribeiro J, Kinnebrew T. Compartment syndromes associated with postoperative epidural analgesia. A case report. J Bone Joint Surg Am. 1996;78(4):597-599.
37. Thonse R, Ashford RU, Williams TI, Harrington P. Differences in attitudes to analgesia in post-operative limb surgery put patients at risk of compartment syndrome. Injury. 2004;35(3):290-295.
38. Whitesides TE Jr. Pain: friend or foe? J Bone Joint Surg Am. 2001;83(9):1424-1425.
39. Staudt JM, Smeulders MJ, van der Horst CM. Normal compartment pressures of the lower leg in children. J Bone Joint Surg Br. 2008;90(2):215-219.
40. Heckman MM, Whitesides TE Jr, Grewe SR, Rooks MD. Compartment pressure in association with closed tibial fractures. The relationship between tissue pressure, compartment, and the distance from the site of the fracture. J Bone Joint Surg Am. 1994;76(9):1285-1292.
41. Hargens AR, Schmidt DA, Evans KL, et al. Quantitation of skeletal-muscle necrosis in a model compartment syndrome. J Bone Joint Surg Am. 1981;63(4):631-636.
42. Heppenstall RB, Sapega AA, Scott R, et al. The compartment syndrome. An experimental and clinical study of muscular energy metabolism using phosphorus nuclear magnetic resonance spectroscopy. Clin Orthop Relat Res. 1988;(226):138-155.
43. McQueen MM, Court-Brown CM. Compartment monitoring in tibial fractures. The pressure threshold for decompression. J Bone Joint Surg Br. 1996;78(1):99-104.
44. Rorabeck CH. The treatment of compartment syndromes of the leg. J Bone Joint Surg Br. 1984;66(1):93-97.
45. Shuler MS, Reisman WM, Kinsey TL, et al. Correlation between muscle oxygenation and compartment pressures in acute compartment syndrome of the leg. J Bone Joint Surg Am. 2010;92(4):863-870.
46. Tobias JD, Hoernschemeyer DG. Near-infrared spectroscopy identifies compartment syndrome in an infant. J Pediatr Orthop. 2007;27(3):311-313.
1. Bhattacharyya T, Vrahas MS. The medical-legal aspects of compartment syndrome. J Bone Joint Surg Am. 2004;86(4):864-868.
2. Prasarn ML, Ouellette EA, Livingstone A, Giuffrida AY. Acute pediatric upper extremity compartment syndrome in the absence of fracture. J Pediatr Orthop. 2009;29(3):263-268.
3. Battaglia TC, Armstrong DG, Schwend RM. Factors affecting forearm compartment pressures in children with supracondylar fractures of the humerus. J Pediatr Orthop. 2002;22(4):431-439.
4. Ramachandran M, Skaggs DL, Crawford HA, et al. Delaying treatment of supracondylar fractures in children: has the pendulum swung too far? J Bone Joint Surg Br. 2008;90(9):1228-1233.
5. Mubarak SJ, Carroll NC. Volkmann’s contracture in children: aetiology and prevention. J Bone Joint Surg Br. 1979;61(3):285-293.
6. Choi PD, Melikian R, Skaggs DL. Risk factors for vascular repair and compartment syndrome in the pulseless supracondylar humerus fracture in children. J Pediatr Orthop. 2010;30(1):50-56.
7. Gupta N, Kay RM, Leitch K, Femino JD, Tolo VT, Skaggs DL. Effect of surgical delay on perioperative complications and need for open reduction in supracondylar humerus fractures in children. J Pediatr Orthop. 2004;24(3):245-248.
8. Iyengar SR, Hoffinger SA, Townsend DR. Early versus delayed reduction and pinning of type III displaced supracondylar fractures of the humerus in children: a comparative study. J Orthop Trauma. 1999;13(1):51-55.
9. Leet AI, Frisancho J, Ebramzadeh E. Delayed treatment of type 3 supracondylar humerus fractures in children. J Pediatr Orthop. 2002;22(2):203-207.
10. Mehlman CT, Strub WM, Roy DR, Wall EJ, Crawford AH. The effect of surgical timing on the perioperative complications of treatment of supracondylar humeral fractures in children. J Bone Joint Surg Am. 2001;83(3):323-327.
11. Diesselhorst MM, Deck JW, Davey JP. Compartment syndrome of the upper arm after closed reduction and percutaneous pinning of a supracondylar humerus fracture. J Pediatr Orthop. 2014;34(2):e1-e4.
12. Mai MC, Beck R, Gabriel K, Singh KA. Posterior arm compartment syndrome after a combined supracondylar humeral and capitellar fractures in an adolescent: a case report. J Pediatr Orthop. 2011;31(3):e16-e19.
13. Blakemore LC, Cooperman DR, Thompson GH, Wathey C, Ballock RT. Compartment syndrome in ipsilateral humerus and forearm fractures in children. Clin Orthop Relat Res. 2000;(376):32-38.
14. Ring D, Waters PM, Hotchkiss RN, Kasser JR. Pediatric floating elbow. J Pediatr Orthop. 2001;21(4):456-459.
15. Haasbeek JF, Cole WG. Open fractures of the arm in children. J Bone Joint Surg Br. 1995;77(4):576-581.
16. Yuan PS, Pring ME, Gaynor TP, Mubarak SJ, Newton PO. Compartment syndrome following intramedullary fixation of pediatric forearm fractures. J Pediatr Orthop. 2004;24(4):370-375.
17. Flynn JM, Jones KJ, Garner MR, Goebel J. Eleven years experience in the operative management of pediatric forearm fractures. J Pediatr Orthop. 2010;30(4):313-319.
18. Blackman AJ, Wall LB, Keeler KA, et al. Acute compartment syndrome after intramedullary nailing of isolated radius and ulna fractures in children. J Pediatr Orthop. 2014;34(1):50-54.
19. Mubarak SJ, Frick S, Sink E, Rathjen K, Noonan KJ. Volkmann contracture and compartment syndromes after femur fractures in children treated with 90/90 spica casts. J Pediatr Orthop. 2006;26(5):567-572.
20. Hope PG, Cole WG. Open fractures of the tibia in children. J Bone Joint Surg Br. 1992;74(4):546-553.
21. Pandya NK, Edmonds EK, Roocroft JH, Mubarak SJ. Tibial tubercle fractures: complications, classification, and the need for intra-articular assessment. J Pediatr Orthop. 2012;32(8):749-759.
22. Flynn JM, Bashyal RK, Yeger-McKeever M, Garner MR, Launay F, Sponseller PD. Acute traumatic compartment syndrome of the leg in children: diagnosis and outcome. J Bone Joint Surg Am. 2011;93(10):937-941.
23. Mubarak SJ. Extensor retinaculum syndrome of the ankle after injury to the distal tibial physis. J Bone Joint Surg Br. 2002;84(1):11-14.
24. Macer GA Jr. Forearm compartment syndrome in the newborn. J Hand Surg Am. 2006;31(9):1550.
25. Ragland R 3rd, Moukoko D, Ezaki M, Carter PR, Mills J. Forearm compartment syndrome in the newborn: report of 24 cases. J Hand Surg Am. 2005;30(5):997-1003.
26. Alioglu B, Avci Z, Baskin E, Ozcay F, Tuncay IC, Ozbek N. Successful use of recombinant factor VIIa (NovoSeven) in children with compartment syndrome: two case reports. J Pediatr Orthop. 2006;26(6):815-817.
27. Lee DK, Jeong WK, Lee DH, Lee SH. Multiple compartment syndrome in a pediatric patient with CML. J Pediatr Orthop. 2011;31(8):889-892.
28. Dumontier C, Sautet A, Man M, Bennani M, Apoil A. Entrapment and compartment syndromes of the upper limb in haemophilia. J Hand Surg Br. 1994;19(4):427-429.
29. Jones G, Thompson K, Johnson M. Acute compartment syndrome after minor trauma in a patient with undiagnosed mild haemophilia B. Lancet. 2013;382(9905):1678.
30. Shaw BA, Hosalkar HS. Rattlesnake bites in children: antivenin treatment and surgical indications. J Bone Joint Surg Am. 2002;84(9):1624-1629.
31. Bae DS, Kadiyala RK, Waters PM. Acute compartment syndrome in children: contemporary diagnosis, treatment, and outcome. J Pediatr Orthop. 2001;21(5):680-688.
32. Noonan KJ, McCarthy JJ. Compartment syndromes in the pediatric patient. J Pediatr Orthop. 2010;30(2 suppl):S96-S101.
33. Dalens B. Some current controversies in paediatric regional anaesthesia. Curr Opin Anaesthesiol. 2006;19(3):301-308.
34. Wedel DJ. Regional anesthesia and pain management: reviewing the past decade and predicting the future. Anesth Analg. 2000;90(5):1244-1245.
35. Mubarak SJ. Wilton NC. Compartment syndromes and epidural analgesia. J Pediatr Orthop. 1997;17(3):282-284.
36. Price C, Ribeiro J, Kinnebrew T. Compartment syndromes associated with postoperative epidural analgesia. A case report. J Bone Joint Surg Am. 1996;78(4):597-599.
37. Thonse R, Ashford RU, Williams TI, Harrington P. Differences in attitudes to analgesia in post-operative limb surgery put patients at risk of compartment syndrome. Injury. 2004;35(3):290-295.
38. Whitesides TE Jr. Pain: friend or foe? J Bone Joint Surg Am. 2001;83(9):1424-1425.
39. Staudt JM, Smeulders MJ, van der Horst CM. Normal compartment pressures of the lower leg in children. J Bone Joint Surg Br. 2008;90(2):215-219.
40. Heckman MM, Whitesides TE Jr, Grewe SR, Rooks MD. Compartment pressure in association with closed tibial fractures. The relationship between tissue pressure, compartment, and the distance from the site of the fracture. J Bone Joint Surg Am. 1994;76(9):1285-1292.
41. Hargens AR, Schmidt DA, Evans KL, et al. Quantitation of skeletal-muscle necrosis in a model compartment syndrome. J Bone Joint Surg Am. 1981;63(4):631-636.
42. Heppenstall RB, Sapega AA, Scott R, et al. The compartment syndrome. An experimental and clinical study of muscular energy metabolism using phosphorus nuclear magnetic resonance spectroscopy. Clin Orthop Relat Res. 1988;(226):138-155.
43. McQueen MM, Court-Brown CM. Compartment monitoring in tibial fractures. The pressure threshold for decompression. J Bone Joint Surg Br. 1996;78(1):99-104.
44. Rorabeck CH. The treatment of compartment syndromes of the leg. J Bone Joint Surg Br. 1984;66(1):93-97.
45. Shuler MS, Reisman WM, Kinsey TL, et al. Correlation between muscle oxygenation and compartment pressures in acute compartment syndrome of the leg. J Bone Joint Surg Am. 2010;92(4):863-870.
46. Tobias JD, Hoernschemeyer DG. Near-infrared spectroscopy identifies compartment syndrome in an infant. J Pediatr Orthop. 2007;27(3):311-313.
Poor sportsmanship
I try to avoid revisiting a subject I have pontificated on in the recent past, but when I encounter a situation in which scientists are behaving unscientifically it is hard to remain silent. In 2002, a Pittsburgh neuropathologist named Bennet Omalu performed an autopsy on Mike Webster, a former National Football League (NFL) lineman who had died in his 50s. Webster had been exhibiting bizarre behaviors and was developing dementia. What Dr. Omalu found in Webster’s brain was a collection of changes that have become known as chronic traumatic encephalopathy (CTE).
In the decade following the publication of Dr. Omalu’s findings in the journal Neurosurgery in 2005, there has been some unsavory back and forths between the NFL’s Mild Traumatic Brain Injury Committee and Dr. Omalu that I learned about in the Wall Street Journal (“The Doctor the NFL Tried to Silence,” by Jeanne Marie Laskas, Nov 24, 2015). The doctor’s side of the story has been published in a book, “Concussion” (New York: Penguin Random House, 2015). “Concussion,” the movie based on the book, was slated for release in December.
The tangle of he said – our experts don’t agree has involved the University of Michigan and Boston University, and the smell of conflict of interest hangs over the NFL’s choice of experts and its decisions to publish or not publish the results of various studies. It now appears that Dr. Omalu’s discovery was the tip of an iceberg of undetermined size. As happens far too often, assumptions and attributions have been made in haste based on scanty evidence from small studies that have surely failed to control for all of the possible contributors.
Considering the results of the autopsies on a few NFL players, it is probably reasonable to suspect that there is something in the culture surrounding professional football that makes some of the players vulnerable to central nervous system damage. And blows to the head are likely to be one of those factors. However, leaping to the conclusion that parents shouldn’t allow their young children to play football is another story. But that is just what Dr. Omalu has done in an op-ed piece that has appeared in the New York Times (“Don’t Let Kids Play Football,” Dec 7, 2015).
Relying heavily on the analogy with cumulative effects of cigarette smoking, Dr. Omalu continues to fan the flame that he ignited with his initial autopsy finding. The timing of the piece is interesting in light of the movie’s release date of Dec. 25. While his discovery of CTE in a professional player is important, Dr. Omalu’s case for prohibiting children from playing football is rife with half-truths and unwarranted conclusions.
For example, he states that in his 30 years as a neuropathologist he has yet to see a “neuron that naturally creates a new neuron to regenerate itself.” True, but he fails to report that there is new evidence that the long-held dictum that neurons can’t heal themselves may be wrong.
Dr. Omalu observes that “if a child who plays football is subjected to advanced radiological and neurocognitive studies during the season and several months after there can be evidence of brain damage at the cellular level even if there were no documented concussions or reported symptoms.” It took some time, but I eventually found the study to which I assume he is referring, by Dr. Christopher T. Whitlow of Wake Forest University, Winston-Salem, N.C., presented at the Radiological Society of North America meeting in December of 2014. Its lead author is careful to state that conclusions should not be drawn from this small preliminary study and observes, “it is unclear whether or not these effects will be associated with any long-term consequences.” However, Dr. Omalu asserts that “If that child continues to play over many seasons, these cellular injuries accumulate to cause irreversible brain damage.” He states this as fact without any supporting evidence.
Fortunately, the American Academy of Pediatrics has presented a more balanced perspective on allowing children to participate in football in light of what we are learning about the health of professional players (“Tackling in Youth Football” [Pediatrics. 2015;136(5)e1419-31]). Dr. William P. Meehan III and Dr. Gregory L. Landry, speaking for the Council on Sports Medicine and Fitness, point out that serious head and neck injury in young football players is very unlikely, and that by teaching proper tackling technique, these injuries can be further decreased.
The real solution to the problem that Dr. Omalu first brought to light in 2002 lies with zero tolerance for the practice of tackling headfirst at all levels of football. Although the NFL has made some feeble attempts to discipline its teams, there is still more that should be done. Every professional and college football game is being video recorded, often from multiple angles. Retrospective analysis of these images should be used to discipline players whose injury-threatening tactics have not been detected by the officials during the game. Multiple game suspensions meted out promptly, and without possibility of appeal, would go a long way to return football to being the safer sport it was when leather helmets discouraged players from using their heads as lethal weapons.
Dr. Wilkoff practiced primary care pediatrics in Brunswick, Maine, for nearly 40 years. He has authored several books on behavioral pediatrics including “How to Say No to Your Toddler.”
I try to avoid revisiting a subject I have pontificated on in the recent past, but when I encounter a situation in which scientists are behaving unscientifically it is hard to remain silent. In 2002, a Pittsburgh neuropathologist named Bennet Omalu performed an autopsy on Mike Webster, a former National Football League (NFL) lineman who had died in his 50s. Webster had been exhibiting bizarre behaviors and was developing dementia. What Dr. Omalu found in Webster’s brain was a collection of changes that have become known as chronic traumatic encephalopathy (CTE).
In the decade following the publication of Dr. Omalu’s findings in the journal Neurosurgery in 2005, there has been some unsavory back and forths between the NFL’s Mild Traumatic Brain Injury Committee and Dr. Omalu that I learned about in the Wall Street Journal (“The Doctor the NFL Tried to Silence,” by Jeanne Marie Laskas, Nov 24, 2015). The doctor’s side of the story has been published in a book, “Concussion” (New York: Penguin Random House, 2015). “Concussion,” the movie based on the book, was slated for release in December.
The tangle of he said – our experts don’t agree has involved the University of Michigan and Boston University, and the smell of conflict of interest hangs over the NFL’s choice of experts and its decisions to publish or not publish the results of various studies. It now appears that Dr. Omalu’s discovery was the tip of an iceberg of undetermined size. As happens far too often, assumptions and attributions have been made in haste based on scanty evidence from small studies that have surely failed to control for all of the possible contributors.
Considering the results of the autopsies on a few NFL players, it is probably reasonable to suspect that there is something in the culture surrounding professional football that makes some of the players vulnerable to central nervous system damage. And blows to the head are likely to be one of those factors. However, leaping to the conclusion that parents shouldn’t allow their young children to play football is another story. But that is just what Dr. Omalu has done in an op-ed piece that has appeared in the New York Times (“Don’t Let Kids Play Football,” Dec 7, 2015).
Relying heavily on the analogy with cumulative effects of cigarette smoking, Dr. Omalu continues to fan the flame that he ignited with his initial autopsy finding. The timing of the piece is interesting in light of the movie’s release date of Dec. 25. While his discovery of CTE in a professional player is important, Dr. Omalu’s case for prohibiting children from playing football is rife with half-truths and unwarranted conclusions.
For example, he states that in his 30 years as a neuropathologist he has yet to see a “neuron that naturally creates a new neuron to regenerate itself.” True, but he fails to report that there is new evidence that the long-held dictum that neurons can’t heal themselves may be wrong.
Dr. Omalu observes that “if a child who plays football is subjected to advanced radiological and neurocognitive studies during the season and several months after there can be evidence of brain damage at the cellular level even if there were no documented concussions or reported symptoms.” It took some time, but I eventually found the study to which I assume he is referring, by Dr. Christopher T. Whitlow of Wake Forest University, Winston-Salem, N.C., presented at the Radiological Society of North America meeting in December of 2014. Its lead author is careful to state that conclusions should not be drawn from this small preliminary study and observes, “it is unclear whether or not these effects will be associated with any long-term consequences.” However, Dr. Omalu asserts that “If that child continues to play over many seasons, these cellular injuries accumulate to cause irreversible brain damage.” He states this as fact without any supporting evidence.
Fortunately, the American Academy of Pediatrics has presented a more balanced perspective on allowing children to participate in football in light of what we are learning about the health of professional players (“Tackling in Youth Football” [Pediatrics. 2015;136(5)e1419-31]). Dr. William P. Meehan III and Dr. Gregory L. Landry, speaking for the Council on Sports Medicine and Fitness, point out that serious head and neck injury in young football players is very unlikely, and that by teaching proper tackling technique, these injuries can be further decreased.
The real solution to the problem that Dr. Omalu first brought to light in 2002 lies with zero tolerance for the practice of tackling headfirst at all levels of football. Although the NFL has made some feeble attempts to discipline its teams, there is still more that should be done. Every professional and college football game is being video recorded, often from multiple angles. Retrospective analysis of these images should be used to discipline players whose injury-threatening tactics have not been detected by the officials during the game. Multiple game suspensions meted out promptly, and without possibility of appeal, would go a long way to return football to being the safer sport it was when leather helmets discouraged players from using their heads as lethal weapons.
Dr. Wilkoff practiced primary care pediatrics in Brunswick, Maine, for nearly 40 years. He has authored several books on behavioral pediatrics including “How to Say No to Your Toddler.”
I try to avoid revisiting a subject I have pontificated on in the recent past, but when I encounter a situation in which scientists are behaving unscientifically it is hard to remain silent. In 2002, a Pittsburgh neuropathologist named Bennet Omalu performed an autopsy on Mike Webster, a former National Football League (NFL) lineman who had died in his 50s. Webster had been exhibiting bizarre behaviors and was developing dementia. What Dr. Omalu found in Webster’s brain was a collection of changes that have become known as chronic traumatic encephalopathy (CTE).
In the decade following the publication of Dr. Omalu’s findings in the journal Neurosurgery in 2005, there has been some unsavory back and forths between the NFL’s Mild Traumatic Brain Injury Committee and Dr. Omalu that I learned about in the Wall Street Journal (“The Doctor the NFL Tried to Silence,” by Jeanne Marie Laskas, Nov 24, 2015). The doctor’s side of the story has been published in a book, “Concussion” (New York: Penguin Random House, 2015). “Concussion,” the movie based on the book, was slated for release in December.
The tangle of he said – our experts don’t agree has involved the University of Michigan and Boston University, and the smell of conflict of interest hangs over the NFL’s choice of experts and its decisions to publish or not publish the results of various studies. It now appears that Dr. Omalu’s discovery was the tip of an iceberg of undetermined size. As happens far too often, assumptions and attributions have been made in haste based on scanty evidence from small studies that have surely failed to control for all of the possible contributors.
Considering the results of the autopsies on a few NFL players, it is probably reasonable to suspect that there is something in the culture surrounding professional football that makes some of the players vulnerable to central nervous system damage. And blows to the head are likely to be one of those factors. However, leaping to the conclusion that parents shouldn’t allow their young children to play football is another story. But that is just what Dr. Omalu has done in an op-ed piece that has appeared in the New York Times (“Don’t Let Kids Play Football,” Dec 7, 2015).
Relying heavily on the analogy with cumulative effects of cigarette smoking, Dr. Omalu continues to fan the flame that he ignited with his initial autopsy finding. The timing of the piece is interesting in light of the movie’s release date of Dec. 25. While his discovery of CTE in a professional player is important, Dr. Omalu’s case for prohibiting children from playing football is rife with half-truths and unwarranted conclusions.
For example, he states that in his 30 years as a neuropathologist he has yet to see a “neuron that naturally creates a new neuron to regenerate itself.” True, but he fails to report that there is new evidence that the long-held dictum that neurons can’t heal themselves may be wrong.
Dr. Omalu observes that “if a child who plays football is subjected to advanced radiological and neurocognitive studies during the season and several months after there can be evidence of brain damage at the cellular level even if there were no documented concussions or reported symptoms.” It took some time, but I eventually found the study to which I assume he is referring, by Dr. Christopher T. Whitlow of Wake Forest University, Winston-Salem, N.C., presented at the Radiological Society of North America meeting in December of 2014. Its lead author is careful to state that conclusions should not be drawn from this small preliminary study and observes, “it is unclear whether or not these effects will be associated with any long-term consequences.” However, Dr. Omalu asserts that “If that child continues to play over many seasons, these cellular injuries accumulate to cause irreversible brain damage.” He states this as fact without any supporting evidence.
Fortunately, the American Academy of Pediatrics has presented a more balanced perspective on allowing children to participate in football in light of what we are learning about the health of professional players (“Tackling in Youth Football” [Pediatrics. 2015;136(5)e1419-31]). Dr. William P. Meehan III and Dr. Gregory L. Landry, speaking for the Council on Sports Medicine and Fitness, point out that serious head and neck injury in young football players is very unlikely, and that by teaching proper tackling technique, these injuries can be further decreased.
The real solution to the problem that Dr. Omalu first brought to light in 2002 lies with zero tolerance for the practice of tackling headfirst at all levels of football. Although the NFL has made some feeble attempts to discipline its teams, there is still more that should be done. Every professional and college football game is being video recorded, often from multiple angles. Retrospective analysis of these images should be used to discipline players whose injury-threatening tactics have not been detected by the officials during the game. Multiple game suspensions meted out promptly, and without possibility of appeal, would go a long way to return football to being the safer sport it was when leather helmets discouraged players from using their heads as lethal weapons.
Dr. Wilkoff practiced primary care pediatrics in Brunswick, Maine, for nearly 40 years. He has authored several books on behavioral pediatrics including “How to Say No to Your Toddler.”
