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Primary Task-specific Bowing Tremor:
An Entity of its Own?
Dr. med. André Lee, MD, and Prof. Dr. med. Eckart Altenmüller, MD, MA A professional violinist in his early 60s, playing in a prestigious or bradykinesia present. Cranial nerves were normal. No rest- German orchestra for more than 20 years, presented to our institute ing tremor was visible on either side. Smelling disorders were because of a task-induced tremor in his right arm when playing the not present.
violin. We describe the phenomenology of this tremor and its treat- ment options and compare it to findings in primary writing tremor Further diagnostic measures including a dopamine trans- (PWT). We then discuss whether primary bowing tremor is an porter (DAT) scan and a cerebral and cervical-spine MRI entity of its own (similar to PWT) and propose hypotheses that were unremarkable. Electrophysiological measures were would derive from such a definition. Med Probl Perform Art 2012; normal for motor evoked potentials (MEP) and marginally prolonged for somatosensory evoked potentials (SEP). In the family history, his father is reported to suffer from either writer's cramp (WC) or primary writing tremor(PWT).
A male violinist in his early 60s had been playing the violin When playing the violin, a pronation-supination tremor for over 50 years, with more than 20 years in a professional as well as a flexion-extension tremor in the wrist of about 7- German orchestra. Tremor appeared first at the age of 46, in 8 Hz was visible and audible (Fig. 1), especially when playing the right arm, while only playing the violin. Prior to this he slow legato notes. Tremor was visible on all four strings of the was involved in a car accident, resulting in a late arrival for a violin equally and was most present between the middle and concert where he had to play numerous, long-lasting, fast, nut of the bow. No postural or resting tremor was visible.
and repetitive bowing-movements (16th notes) in a long-last- When the left hand was used for bowing, no tremor ing, romantic symphony. The following day he developed a pain syndrome in the right shoulder. Subsequently, he rec- Tremor appeared in everyday situations (e.g., filling a glass ognized a progressive involuntary tremor of the right arm of water) about 1.5 years ago, however to a lesser extent and while playing the violin. From 2005 onward, he has received unilateral as well.
medical treatment for continued pain syndromes in the mus- Tremor initially responded to beta-blockers (propranolol culoskeletal system. 20 mg); however, this effect wore off over the course of 1 year.
In 2010, he presented to our institute for the first time Alcohol still improves symptoms. Trihexiphenidyl treatment- because of accelerated tremor progression for 1 year, which did not lead to an improvement. Injections of botulinum had caused him to reduce or cancel solo- or chamber music toxin (Dysport®) were given twice and had a positive effect in everyday life activities and some effect when playing the Clinical neurological examination did not reveal any violin, without side effects. The first injections were given to pathologic findings. Reflexes were symmetrical without the bicep, tricep, pronator teres, and supinator muscles. The pathologic reflexes. No sensory deficit was detectable. Pal- second injections were localized to the radial extensor carpi, laesthesia was normal. Diadochokinesis was undisturbed.
radial flexor carpi, supinator, bicep, and tricep muscles.
There were no signs of cerebellar dysfunctions and no rigor Treatment was not continued, because improvement at theviolin was insufficient.
Primidone (anticonvulsant) led to an improvement, but Lee is Postdoctoral Research Fellow and Altenmüller due to side effects (tiredness, slowing, and erectile dysfunc- is Director at the University of Music, Drama and Media Hannover, and tion), continuous treatment had to be stopped. He currently the Institute for Music Physiology and Musicians' Medicine, Hannover, takes primidone 75 mg prior to playing solos or chamber music. Neither gabapentin nor topiramate (anticonvulsants) Address correspondence to: Dr. André Lee, University of Music, Drama and was effective. Clozapine was proposed, but so far not taken.
Media Hannover, Institute for Music Physiology and Musicians' Medicine, Deep brain stimulation was declined by the patient.
Emmichplatz 1, 30175 Hannover, Germany. Tel +49 511 3100 552, fax The patient continues to play in the orchestra and has +49 51 3100 557.
again started to perform chamber music concerts, however 2012 Science & Medicine.
less frequently than before tremor onset.
224 Medical Problems of Performing Artists
Tremor spread to other tasks about 14 years after onset of primary bowing tremor. However, this did not affect hiseveryday life, and tremor at the violin remained the mostprominent and disturbing symptom. Tremor spreading toother tasks has been described in PWT before,4,11 and a stricttask-specificity was thus negated.10,11 Rather, an evolution ofthe disorder at different points in time11 was discussed. Family history was positive, with the father suffering from either WC or PWT. This alludes to a genetic component inbowing tremor that has been described in PWT.4,11 Environmental factors may play a role in primary bowing tremor as well as in PWT. Bain et al.4 showed that 19% ofpatients with PWT had a previous history of trauma. Thiscorresponds to our patient, who had been involved in a caraccident with a subsequent pain syndrome of the right armprior to tremor onset. Our treatment included botulinum toxin, which led to a slight improvement. An improvement of tremor after botu-linum toxin injection also has been reported in PWT.12 Oral medications included propranolol, primidone, gabapentin and trihexiphenidyl, of which most effective wereprimidone and propranolol. Trihexiphenidyl had a slight FIGURE 1. Rectified EMG of the flexor carpi radialis (upper panel)
effect. This is in accordance with reports that about 33% of and the extensor carpi radialis (lower panel) with a reciprocal activity patients with PWT respond specifically to primidone or pro- of about 7–8 Hz.
pranolol.4 Alcohol, which also has been reported to improvesymptoms in about 33% of PWT-patients,4 also relieved tremor in our patient.
Tremor is defined as a rhythmical, oscillatory, and involun- From a phenomenological point of view, our patient's symp- tary movement of a body part1 and is one of the most toms are very similar to those displayed in patients with common movement disorders. Task-specific tremors have PWT. Both tremors occur in a highly trained fine motor task.
been defined in the consensus statement on tremor in 1998.1 We therefore hypothesize that primary bowing tremor (PBT) The most common task-specific tremor is PWT, which was and PWT may share similar underlyingpathophysiological first described by Rothwell in 1979.2 There are two types of mechanisms so that PBT may be an entity of its own, as has PWT: type A, which refers to tremor induced by writing, and been proposed by Lederman13 (2010). type B, which refers to tremor induced when holding the arm The main pathophysiologic findings in PWT that corrob- in the position for writing.2 orate the definitions of PWT as an entity of its own are: It has since been an ongoing discussion, whether PWT is a type of focal dystonia, a local form of essential tremor, or a 1. No excessive overflow in EMG recordings in PWT as opposed to nosological entity of its own.1,3–6 Recent research suggests that it is rather a separate entity.5–10 2. Normal reciprocal inhibition in PWT4,8 as compared to WC.14,153. In a recent study by Meunier et al.,16 TENS was shown to have a Primary Bowing Tremor and PWT
deteriorative effect in PWT 6, whereas in WC high-frequencyTENS may be beneficial. We report on a violinist in his early 60s, who presented to 4. Normal results for spinal and motor cortex excitability8 were our institute because of a unilateral tremor. The tremor was obtained in PWT, whereas it may be abnormal in essentialtremor and is abnormal in WC.17,18 present in the right arm and induced by playing the violin.
Age of onset was 46. He fulfilled the three criteria for focal Defining PBT as an entity of its own, similar to PWT, has task-specificity as described by Rosenbaum and Jankovic3: a) the advantage in that it will allow a systematic approach to tremor was unilateral and limited to one body part, i.e., the new hypotheses concerning the pathophysiology of PBT: right arm (focal); b) no other movement disorders were pres-ent; and c) tremor was at first provoked only by playing the 1. PBT shows no excessive overflow of EMG activity.
violin (task specific), corresponding to a type A tremor. It was 2. PBT show normal reciprocal inhibition.
mainly a pronation-supination tremor, similar to the move- 3. TENS does not have a beneficial effect on PBT.
ment described in PWT by Rothwell.2 4. PBT has a normal intracortical and spinal excitability.
December 2012 225
With regard to treatment options, we would hypothesize 10. Bain PG. Task-specific tremor. Handb Clin Neurol 2011; 100:711–718.
that thalamic stimulation may have a beneficial effect on PBT, Ondo WG, Satija P. Task-specific writing tremor: clinical phenotypes, as has been shown for PWT.19,20 Furthermore, brain imaging in progression, treatment outcomes, and proposed nomenclature. Int JNeurosci 2012; 122(2):88-91. doi:10.3109/00207454.2011.630544. PBT should reveal a distinct activation pattern similar to that in 12. Papapetropoulos S, Singer C. Treatment of primary writing tremor PWT.7 Future research is needed to clarify these hypotheses.
with botulinum toxin type a injections: report of a case series. Clin Neu-ropharmacol 2006; 29:364–367.
13. Lederman RJ: The tremulous bow arm: technical or neurological? Pre- sented at the 28th Annual Symposium of Medical Problems of Per- 1. Deuschl G, Bain P, Brin M. Consensus statement of the Movement forming Artists, 2010, Aspen Colorado.
Disorder Society on Tremor. Ad Hoc Scientific Committee. Mov.
14. Nakashima K, et al. Reciprocal inhibition between forearm muscles in Disord. 1998; 13 (suppl 3):2–23.
patients with writer's cramp and other occupational cramps, sympto- 2. Rothwell JC, Traub MM, Marsden CD. Primary writing tremor. J matic hemidystonia and hemiparesis due to stroke. Brain 1989; 112 (pt Neurol Neurosurg Psychiatr 1979; 42:1106–1114.
3. Rosenbaum F, Jankovic J. Focal task-specific tremor and dystonia: cat- 15. Panizza M, Lelli S, Nilsson J, Hallett M. H-reflex recovery curve and egorization of occupational movement disorders. Neurology 1988; reciprocal inhibition of H-reflex in different kinds of dystonia. Neurol- ogy 1990; 40:824–828.
4. Bain PG, et al. Primary writing tremor. Brain 1995; 118(pt 16. Tinazzi M, et al. Effects of transcutaneous electrical nerve stimulation on motor cortex excitability in writer's cramp: Neurophysiological and 5. Hai C, Yu-ping W, Hua W, Ying S. Advances in primary writing clinical correlations. Move Disord 2006; 21:1908–1913.
tremor. Parkinsonism Relat Disord 2010; 16:561–565.
Ridding MC, Sheean G, Rothwell JC, et al. Changes in the balance 6. Meunier S, et al. TENS is harmful in primary writing tremor. Clin Neu- between motor cortical excitation and inhibition in focal, task specific rophysiol 2011; 122:171–175.
dystonia. J Neurol Neurosurg Psychiatr 1995; 59:493–498.
7. Berg D, Preibisch C, Hofmann E, Naumann M. Cerebral activation 18. Chen R, Wassermann EM, Caños M, Hallett M. Impaired inhibition pattern in primary writing tremor. J Neurol Neurosurg Psychiatr 2000; in writer's cramp during voluntary muscle activation. Neurology 1997; 8. Modugno N, et al. Neurophysiological investigations in patients with 19. Minguez-Castellanos A, et al. Primary writing tremor treated by primary writing tremor. Mov Disord 2002; 17:1336–1340.
chronic thalamic stimulation. Mov Disord 1999; 14:1030–1033.
9. Espay AJ, Chen R. Primary writing tremor and writer's cramp: distinct 20. Racette BA, Dowling J, Randle J, Mink JW. Thalamic stimulation for phenomenology, diverging pathophysiology. Clin Neurophysiol 2011; primary writing tremor. J Neurol 2001; 248:380–382.
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