Dystonia and the Long Distance Runner
Abstract & Commentary
By Claire Henchcliffe, MD, DPhil, Assistant Professor, Department of Neurology, Weil Medical College, Cornell University. Dr. Henchcliffe reports no financial relationship to this field of study.
Synopsis: This case series describes clinical features and treatment responses of an unusual focal leg action dystonia in 5 long distance runners.
Source: Wu LJ and Jankovic J. Runner's Dystonia. J Neurol Sci. 2006;251:73-76.
The authors describe 5 unusual cases of focal leg and/or foot dystonia occurring in long distance runners seen at an academic movement disorders center. Mean age at symptom onset was 37.4 ± 10.33 years (range 20-46 years), and mean symptom duration at the time of evaluation was 7.2 ± 4.44 years (range 2-12 years). Three of the 5 subjects were women. Four of the 5 had foot flexion or inversion as a presenting symptom, and in 3 of these it spread to involve knee extension, with hip flexion in one. In one subject, only the distal limb was involved. In all cases, symptoms were initially present only when running, and triggers later identified included walking on tip toes, squatting, cycling > 20 minutes, walking up stairs, and running wearing shoes. Three found sensory tricks to relieve dystonia, including walking backwards, walking on heels, and touching the hip. Two subjects exhibited a dystonic tremor with typical null point, that is, relief with certain postures. Only 2 of the cases are described in detail, and both had a prior injury, superficial to both knees after tripping in one (2 months prior to symptoms), and knee injury with swelling from a motor vehicle accident followed by patellar tendon transfer (10 months prior to symptoms) in the other. In all 5 cases, brain MRIs and EMG/NCV studies were normal, there was no family history of dystonia, and DYT1 gene testing was negative in the 3 cases in which it was performed. Patients responded to botulinum toxin A injections (n = 2), levodopa (n = 1), carbamazepine (n = 1), trihexiphenidyl (n = 1), but no details are available as to how many other medications were tried in each case.
Commentary
Focal adult-onset dystonias most commonly involve blepharospasm, torticollis, and oromandibular dystonia. Isolated adult-onset foot dystonia is often a red flag that the patient will go on to develop Parkinson's disease or another parkinsonian disorder. However, the unusual pattern of dystonia described here, with an isolated limb presentation, and association with voluntary motor activity, fits with other focal action dystonias such as writer's cramp, musician's dystonia,1 and (likely) golfers' "yips." Moreover, the authors have adequate time periods of follow up without parkinsonian symptoms developing. Such disorders can be challenging to recognize, leading to misdiagnosis in some; of note, one of the above cases was seen by 2 neurologists, a psychiatrist, and 3 orthopedic surgeons prior to diagnosis. In particular, the paroxysmal nature of the movements, and somewhat bizarre phenomenon of sensory tricks, can lead to the impression of a psychogenic disorder. The range of treatments used successfully by the authors is encouraging, but there is clearly much to learn. In particular, the authors caution that if mistaken for an orthopedic disorder, casting could be recommended, and this could be detrimental based upon case reports of dystonia onset after limb immobilization. However, there are also reports of treatment of limb dystonia involving immobilization. Finally, as the authors point out, a prior history of limb injury in 2/5 cases raises the question of whether these could be peripherally induced movement disorders. Overall, this small case series highlights a fascinating and poorly understood disorder, and will hopefully enhance its recognition in other individuals, as well as stimulate further study.
Reference:
1. Frucht SJ. Focal Task-Specific Dystonia in Musicians. Adv Neurol. 2004;94:225-230.
This case series describes clinical features and treatment responses of an unusual focal leg action dystonia in 5 long distance runners.You have reached your article limit for the month. Subscribe now to access this article plus other member-only content.
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