Focal hand dystonia in musicians: phenomenology, etiology, and psychological trigger factors

½Q1

ARTICLE IN PRESS

123456789

10111213141516171819202122232425262728293031323334353637383940414243444546474849505152535455565758

Focal hand dystonia in musicians:phenomenology, etiology, and psychological

trigger factors

596061626364656667

Eckart AltenmullerUniversity for Music and Drama, Hannover Institute forMusic Physiology and Musicians’ Medicine,Hohenzollernstrasse 47, D-30161 Hannover, Germany

Hans-Christian JabuschInstitute of Musicians’ Medicine, Dresden University ofMusic ‘‘Carl Maria von Weber,’’ Dresden,� Germany

Correspondence and reprint requests to Tel.: þ49 511 3100 552; fax:þ49 511 3100 557; e-mail: <[email protected]>.

0894-1130/$ e see front matter � 2008 Hanley & Belfus, an imprintof Elsevier Inc. All rights reserved.

doi:10.1016/j.jht.2008.11.007

686970717273747576

FLA 5.0 DTD � HANTHE511_proof

ABSTRACT: Musician’s dystonia is a task-specific movementdisorder, which manifests itself as a loss of voluntary motor controlin extensively trained movements. In many cases, the disorderterminates the careers of affected musicians. Approximately 1%of all professional musicians are affected. In the past, focal dystonia(FD) was classified as a psychological disorder. Over time, theproblem was classified as a neurological problem. Although thespecific pathophysiology of the disorder is still unclear, it appearsthe etiology is multifactorial. While there may be a genetic history,neurophysiological, physical, and environmental factors, traumaand stress contribute to the phenotypic development of FD.

Are there potentially psychological factors that also contribute tothe manifestation of the disorder? This manuscript analyzes theevidence supporting the potential contribution of the emotionalbrain systems in the etiology of focal hand dystonia in musicians.In addition, the psychological findings from a large descriptivestudy comparing healthy musicians, musicians with dystonia,and musicians with chronic pain (CP) are presented. Informationabout psychogenic characteristics might be used to modify inter-vention strategies and music instruction to reduce the incidenceof musician’s dystonia.

J HAND THER. 2008;jj:jej.

7778798081828384858687888990919293949596979899

WHAT IS MUSICIAN’S DYSTONIA?

Focal dystonia in musicians, also known as musi-cian’s cramp or musician’s dystonia, is a task-specificmovement disorder, which presents itself as muscu-lar incoordination or loss of voluntary motor controlof extensively trained movements while a musician isplaying the instrument. For those who are affected,FD is highly disabling and in many cases terminatesmusical careers.1

Musician’s dystonia may be classified according tothe task specifically involved. For example, embou-chure dystonia may affect coordination of lips,tongue, facial and cervical muscles and breathing inbrass and wind players, whereas pianist’s cramp andviolinist’s cramp affect the control of hand move-ments. According to recent estimates, 1% of allprofessional musicians are affected.1 In contrast, inthe general population, prevalence of FDs, includingwriter’s cramp, blepharospasm, and cervical dysto-nia, is estimated as 29.5/100,000 people in the

United States and 6.1/100,000 people in Japan.2,3 Incomparison to other activities producing dystonicmovements, such as writing, playing golf (the‘‘yips’’) or dart (‘‘dartism’’), classical musicians areat the highest risk of developing FD.

Typically, musician’s dystonia occurs without pain,although muscle aching can present after prolongedspasms. Lack of pain distinguishes it from repetitivestrain injury or occupational fatigue syndrome. It isimportant to make this distinction bearing in mindthat prolonged pain syndromes may lead to symp-tomatic dystonia, possibly due to the degradation ofsensory receptive fields in the somatosensory cortex.The loss of muscular coordination is frequentlyaccompanied by a co-contraction of antagonist mus-cle groups. For example in pianist’s cramp, the co-activation of wrist flexor and wrist extensor musclesis frequently observed. In Figure 1, typical postures ofdystonic finger movements in musicians are shown.

100101102103104

WHAT CAUSES MUSICIAN’SDYSTONIA?

Until today, the etiology of musician’s dystonia hasnot been completely understood, but it is probablymultifactorial. It may develop in individuals with a

jej 2008 1

� 26 December 2008 � 8:29 pm

mailto:[email protected]

FIGURE 1. Typical patterns of dystonic posture in a pianist, a violinist, a flutist, and a trombone player.

ARTICLE IN PRESS

105106107108109110111112113114115116117118119120121122123124125126127128129130131132133134135136137138139140141142143144145146147148149150151152153154155156157158159160161162

163164165166167168169170171172173174175176177178179180181182183184185186187188189190191192193194195196197198199200201202203204205206207208209210211212213214215216217218219220

genetic history of dystonia4 or as a consequence ofalterations in the basal ganglia circuitry.5,6 Most stud-ies of musician’s dystonia reveal abnormalities inthree main areas: a) reduced inhibition in the motorsystem at cortical, subcortical, and spinal levels b)reduced sensory perception and integration; and c)impaired sensorimotor integration. The latterchanges are mainly believed to originate from dys-functional brain plasticity. Such a dysfunctionalplasticity has been described in the sensory thala-mus.7 Disorganized motor somatotopy could befound in the putamen of patients suffering fromwriters’ cramp.8 Finally, there is growing evidencefor abnormal cortical processing of sensory informa-tion as well as degraded representation of motorfunctions in patients with FD. In monkeys, repetitivemovements induced symptoms of focal hand dysto-nia and a distortion of the cortical somatosensoryrepresentation,9 suggesting that practice-inducedalterations in cortical processing may play a role infocal hand dystonia.

Indeed, using somatosensory-evoked potentialtechnology, it was demonstrated that in the somato-sensory cortex the topographical location of sensoryinputs from individual fingers overlap more inpatients with writer’s cramp than in healthy con-trols.10 Similar observations have been made usingmagnetoencephalography.11 Elbert et al.12 showedthat there is an overlap of the representational zonesof the digits in the primary somatosensory cortex forthe affected hand of musicians with dystoniacompared with the representations of the digits innonmusician control subjects.

2 JOURNAL OF HAND THERAPY


In healthy musicians, an increase in sensory fingerrepresentations has been described and interpretedas adaptive plastic changes to conform to the currentneeds and experiences of the individual.13 It could bespeculated that these changes develop too far inmusicians suffering from dystonia, shifting brainplasticity from a beneficial to a mal-adaptationlevel.14 On the behavioral level, this notion is sup-ported by the fact that healthy musicians have low-ered two-point discrimination thresholds comparedwith nonmusician controls,15 whereas two point-discrimination thresholds are increased in musicianssuffering from dystonia.

Another clinical sign, which emphasizes the im-portant role of sensorimotor integration in the path-ophysiology of musician’s dystonia, is the ‘‘sensorytrick’’ phenomenon. This phenomenon is knownfrom patients with cervical dystonia: touching theface contralateral, but also ipsilateral to the directionof head, rotation can reduce or abolish involuntarymuscle activity.16 In a similar way, musicians suffer-ing from dystonia frequently experience markedimprovement of fine motor control when playingwith a latex glove, or when holding an object, forexample a rubber gum, between the fingers, thuschanging the somatosensory input information.17

Interestingly, in musicians with hand dystonia, anassociation exists between the instrument group andthe localization of FD. In instruments with differentwork loads, different complexity of movements, ordifferent temporospatial precision for both hands, FDappears more often in the more heavily used hand.Keyboard musicians (piano, organ, harpsichord) and

� 26 December 2008 � 8:29 pm

ARTICLE IN PRESS

221222223224225226227228229230231232233234235236237238239240241242243244245246247248249250251252253254255256257258259260261262263264265266267268269270271272273274275276277278

279280281282283284285286287288289290291292293294295296297298299300301302303304305306307308309310311

those with plucked instruments (guitar, e-bass) areprimarily affected in the right hand. All these instru-ments are characterized by a higher workload in theright hand. Additionally, guitar playing requireshigher temporospatial precision in the right handcompared with that in the left hand. Bowed stringplayers who have a higher workload and complexityof movements in the left hand are predominantlyaffected in the left hand.1

In summary, in view of neurophysiological andepidemiological findings in musicians with FD, sev-eral predisposing factors have been identified, suchas male gender,18,19 as well as a positive family his-tory,20 which might constitute a particular vulnerabil-ity or predisposition to musician’s dystonia.Additional extrinsic and intrinsic factors may triggerthe manifestation of musician’s dystonia on the basisof a given predisposition. Intrinsic triggering factorsare, for example, physical disorders resulting in localpain and/or intensified somatosensory input.Traumatic injuries, nerve-entrapment, or overuse in-jury may also lead to a degradation of sensorimotorrepresentations at several levels of the sensorimotorcircuits.21

Extrinsic triggering factors, according to epidemi-ological findings, 22e24 are spatial and temporal sen-sorimotor constraints as well as musical and socialconstraints typical of the performance situation inclassical music. The question remains open, whetherpsychological factors contribute to the manifestationof the disorder. In this manuscript, we review the lit-erature on the potential role of psychological factorsin musician’s dystonia.

312313314315316317318319320321322323324325326327328329330331332333334335

ARE PSYCHOLOGICAL FACTORSINFLUENCING AND TRIGGERINGDYSTONIA?

For many decades, psychological factors werebelieved to be essential for the development of task-specific FDs and were overemphasized in the as-sumed pathomechanism. Gowers subsumed writer’scramp and related cramps under the term ‘‘occupa-tional neuroses.’’25 At that time, ‘‘neurosis’’ was usedas a term for a disease when a physical origin wasassumed, but a clear cause could not be described.As a consequence of the terminology, a psychogenicorigin was assumed (for example: Ref. 26) until in1982 Sheehy and Marsden postulated a neurologicalpathomechanism, because psychiatric investigationof 29 patients with writer’s cramps and relatedcramps (four with typist’s cramps, one with pianist’scramp) did not reveal a higher incidence of psychiat-ric disturbance than that in a normal population, asjudged by formal Present State Examination.27 Theystated that occupational cramps are symptoms of aphysical illness, and they used the term ‘‘FD.’’28


Subsequently, the neuropathophysiology of FD wasinvestigated systematically and yielded the abovebriefly reviewed results.

At the same time, a number of studies pointed atpsychological findings in patients with writer’scramp and emphasized a psychological and psycho-social part in the etiology of the disorder.29,30

Windgassen and Ludolph found depressive, anan-castic, sensitive and hysteric traits in 22 patientswith writer’s cramp.31 The Giessen Test showed aninclination toward compulsive traits in these pa-tients. Anxiety was occasionally observed in patientswith writer’s cramp.32e34 It was discussed controver-sially whether these special psychological conditionsare part of the etiology of FD35 or whether they dem-onstrate secondary psychoreactive processes.36 Incontrast, in a study with 22 patients suffering fromwriter’s cramp, no significant differences were foundcompared to a matched normal control group usingthe Crown-Crisp Experiential Index.37 This question-naire assessed traits and symptoms relevant to neu-rotic illness and contained three anxiety subscales(free-floating anxiety, phobic anxiety, somatic anxi-ety). Gundel et al.38 compared psychiatric comorbid-ity in patients suffering from spasmodic dysphonia, aFD of the voice impairing speaking as well as singing,and compared it to patients with vocal fold paresis.Surprisingly, 41.7% of patients with spasmodic dys-phonia, compared with 19.5% of patients with vocalfold paresis, had a current psychiatric diagnosis.Depression, anxiety, and adjustment disorders werethe prevailing symptoms in both groups.

Recent studies have revealed additional, thoughsubtle, behavioral dysfunctions in patients with FD.Bugalho and colleagues39 demonstrated increasedperseveration in the Wisconsin Card Sorting Test,indicative of defective set shifting, and higher inten-sity of obsessive compulsive disorder in patientswith FD. These findings may reflect a pattern of com-plex neurophysiological dysfunction involving dor-solateral, orbitofrontal, and motor frontostriatalcircuits. In fact, in an electroencephalograph studyin pianists, suffering from dystonia, electrophysio-logical correlates of such a defective set shifting couldbe convincingly demonstrated. In comparison withhealthy pianists, those with dystonia could not ap-propriately deactivate the preparatory cortical activa-tion in anticipation of playing a C-major scale, when aNo-go condition was introduced requiring them notto play the scale.40

THE HANNOVER STUDY:PSYCHOLOGICAL FINDINGS INMUSICIAN’S DYSTONIA

All the above mentioned psychological studieswere undertaken in nonmusician patients suffering

jej 2008 3

336

� 26 December 2008 � 8:29 pm

ARTICLE IN PRESS

337338339340341342343344345346347348349350351352353354355356357358359360361362363364365366367368369370371372373374375376377378379380381382383384385386387388389390391392393394

395396397398399400401402403404405406407408409410411412413414415416417418419420421422423424425426427428429430431432433434435436437438439440441442443444445446447448449450451452

from various forms of FDs. It is intriguing that newfindings point at a different origin of musician’sdystonia compared with other FDs.41 In general, itseems that musician’s dystonia is more linked tooveruse and overpractice compared with otherhand dystonias.

In this context it is worth mentioning that musi-cian’s dystonia emerged relatively recently duringthe early romantic period, when eminent virtuosossuch as Paganini or Franz Liszt pushed technicaldemands to new limits. The first proven records ofthis condition date back only to 1830, when theambitious pianist and composer Robert Schumanndeveloped a pianist’s cramp, deteriorating the finemotor control of the middle finger of his right hand.42

As a supposedly precipitating factor, RobertSchumann had dramatically increased his pianopractice schedules up to 7 hrs a day to attain the pia-nistic technique necessary to compete with the emi-nent virtuosos of the early romantic period.However, besides prolonged practice time and mus-culoskeletal overuse, psychosocial stressors and per-sonality factors may have contributed to his disorder.According to his diaries and the written testimonialsof his friends, Schumann tended to compulsive work-ing behavior, to harsh self-criticism, to anxiety anddepression, and to frequent and excessive alcoholconsumption. Already at this time, his physiciansdiscussed whether he presented a psychogenic or aneurological condition.43

The Hannover study on psychologicalconditions in musicians

We have investigated personality structures inmusicians with FD.42,43 This study was designed toexamine psychological conditions in musicians withFD and to compare them with healthy musiciansand those suffering from CP syndromes. The lattergroup was added to detect unspecific secondary psy-chological reactions in diseased musicians. Based onclinical observations, the underlying hypothesis wasthat a) musicians with dystonia more often sufferfrom social phobias and specific phobias and displaymore perfectionistic tendencies than healthy musi-cians and those with CP and that b) these psycholog-ical conditions were already present before onset ofdystonia.

Two groups of patients were included: One samplecomprised 20 professional musicians who had beendiagnosed with task-related FDs. Three out of 20patients were brass players with embouchure dysto-nias; the other 17 patients suffered from hand dysto-nias, which presented in the typical manner aspainless cramping of one or more fingers while thepatients played their instruments. The individualdurations of the disorder were between 3 and 12years (6.9 6 2.6 years [mean 6 SD]) at the time of the



study. Twelve of the patients noticed additional dys-tonic movement patterns in other activities such aswriting (five patients), on the computer keyboard(four patients), or everyday activities; the onset ofthese additional symptoms took place after the onsetof musicians cramps. Patients with other neurologi-cal disorders or secondary dystonias were excludedfrom the study.

The second sample of patients comprised 20 pro-fessional musicians suffering from CP syndromesrelated to playing their instruments. Pain was local-ized in the hand and/or arm region. The durations ofCP ranged from 6 months to 30 years (4.9 6 6.9 years)at the time of the study.

A third group consisting of 30 professional musi-cians was recruited as healthy controls. They wereworking in German orchestras, Colleges of Music,and as freelance professional musicians. By means ofquestionnaires, they were asked if they were suffer-ing from any kind of ill health, especially frommovement disorders or CP. Musicians with anysomatic complaints or neurological or psychiatricdiseases were excluded from the normal controlsample. The samples of patients and the controlgroup were similar in age. However, both patientgroups differed markedly in gender distributions.There was a preponderance of males (16 males, fourfemales) in the sample of musicians with FDs,whereas females were predominant in the groupwith CP (five males, 15 females). The respectivegender distributions are in keeping with reports ofother authors who found more male than femalemusicians suffering from FD46 and a preponderanceof females in patients with CP.47 Gender distributionwas almost balanced in the sample of normal controlswith 16 males and 14 females. It should be mentionedthat the process of finding subjects for the healthycontrol group was protracted due to the high numberof symptomatic musicians among the professionals.This observation coincides with the results ofFishbein et al., who found that 76% of orchestra mu-sicians reported at least one medical problem thatwas severe in terms of its effect on performance.49

Methods of the study

The assessment of psychological conditions wasbased on self-estimation using the following Germaninventories: the revised version of the FreiburgPersonality Inventory (FPI-R)49 is a personality ques-tionnaire, which consists of 12 bipolar subscales be-ing life satisfaction, social orientation, achievementorientation, inhibitedness, irritability, aggressiveness,strain, somatic complaints, health concern, frankness,extraversion, and emotionality. Additionally, theQuestionnaire for Competence and ControlOrientations (QCC)50 was used to investigate fea-tures such as self-concept of abilities, internal control

� 26 December 2008 � 8:29 pm

FIGURE 2. Findings of the anxiety questionnaire of con-trols and musicians with chronic pain and those with focaldystonia: percentage of subjects (of respective groups) whoreported any anxiety disorder (A), social phobia (B), spe-cific phobias (C), free-floating anxiety (D). Hatched bars:percentage with anxiety before onset of disorder. Filledbars: percentage with anxiety after onset of disorder(*p , 0.05, **p , 0.01, n.s.: not significant).

ARTICLE IN PRESS

453454455456457458459460461462463464465466467468469470471472473474475476477478479480481482483484485486487488489490491492493494495496497498499500501502503504505506507508509510

511512513514515516517518519520521522523524525526527528529530531532533534535536537538539540541542543544545546547548549550551552553554555556557558559560561562563564565566567568

orientation, others control orientation, and chancecontrol orientation (primary subscales). In order toassess perfectionistic tendencies, a questionnairewas developed with five items on a six-point scale.Finally, six questions were designed focusing on anx-iety disorders subgrouped as considerable stagefright, panic attacks, free-floating anxiety, agorapho-bia, social phobia, and specific phobias such as acro-phobia, claustrophobia, and so on. The respectivesymptoms were explained to the subjects, and theywere asked whether they felt that these anxietieswere present or absent. To investigate whether cer-tain psychological conditions were already presentbefore the onset of FD or CP in the patient groups,a second step was added: subjects were asked todecide whether the particular anxieties had beenpresent or absent before onset of their disorders,and whether their attitudes concerning perfectionismwere the same or different before and after onset.

Informed consent was obtained from all subjects.They were instructed to fill out the questionnaireswithout the presence of other persons. Distributionand collection of the questionnaires were done bymailing, and precise written explanations were at-tached. Questionnaires that were not filled out prop-erly were excluded from the study. FPI-R and QCCquestionnaires with incomplete data were treatedfollowing the rules for missing data treatment rec-ommended by the authors of the questionnaires.Data of subjects who showed dissimulative tenden-cies in the frankness subscale of the FPI-R were notincluded in the study.

Statistical analyses were performed (STATISTICAVersion 5.5 A, StatSoft Inc.): Differences betweengroups and gender concerning FPI-R, QCC (primarysubscales), and the perfectionism scale were calcu-lated using multivariate analyses of variances(MANOVA). Statistical analyses of the differencesin anxiety disorders between the groups were per-formed using chi-square test (x2). Two-tailed Fisher’sexact tests were applied when x2-tests were not pos-sible due to methodological requirements. x2-tests/Fisher’s exact tests were performed separately forevaluation of the differences in the anxiety subscalesbefore and after onset of the disorders.

Results of the study

Anxieties were present more often in both patientgroups (FD and CP) than in normal controls (FD:x2¼ 5.33, p , 0.05; CP: x2¼ 9.07, p , 0.01) as shownin Figure 2.

Patients were asked to specify whether their ten-dencies concerning anxieties had been the same ordifferent before and after the onset of their diseases.In this respect, conclusive statements could beobtained in all patients with FD and in 19 out of 20patients with CP. x2-tests/Fisher’s exact tests were


performed separately for evaluation of the differ-ences in the anxiety subscales before and after onsetof the disorders. Significantly, in both patient groupsanxieties had been more often present already beforeonset of the disorders compared with healthy musi-cians (FD: x

2¼ 5.33, p , 0.05; CP: x2¼ 6.38,p , 0.05). A closer look at the different anxiety sub-scales showed that musicians with FD suffered

jej 2008 5

� 26 December 2008 � 8:29 pm

½Q2�

ARTICLE IN PRESS

569570571572573574575576577578579580581582583584585586587588589590591592593594595596597598599600601602603604605606607608609610611612613614615616617618619620621622623624625626

627628629630631632633634635636637638639640641642643644645646647648649650651652653654655656657658659660661662663664665666667668669670671672673674675676677678679680681682683684

from social phobias remarkably more often thanhealthy musicians (two-tailed Fisher’s exact test(Fisher’s), p , 0.01) which had already been thecase before onset of FD (Fisher’s, p , 0.01). Patientswith CP did not report more social phobias presentalready before onset of CP compared with healthymusicians. However, this comparison revealed asignificant difference (Fisher’s, p , 0.05) after devel-opment of CP due to an increase in social phobiasin these patients after onset. Only patients with FDsignificantly more often reported specific phobiassuch as acrophobia, claustrophobia, and so on, com-pared with healthy musicians (x2¼ 4.69, p , 0.05).These specific phobias had already been presentbefore they developed FD. Musicians who laterdeveloped FD suffered markedly more often fromspecific phobias than musicians who later developedCP (Fisher’s, p , 0.05). The opposite result was foundin the subscale free-floating anxiety: only patientswith CP significantly more often reported free-floating anxiety than normal controls. This was thecase before (x2¼ 5.21, p , 0.05) and after onset ofCP (x2¼ 8.0, p , 0.01). In the subscales agoraphobia,panic attacks, and remarkable stage fright, no differ-ences were observed between any patient group andhealthy musicians. No significant differences werefound in any of the anxiety subscales between maleand female subjects.

Using 3 (group affiliation) 3 2 (gender) MANOVAfor the perfectionism questionnaire and all subscalesof the QCC and FPI-R, significant differences wereseen between the groups FD, CP, and control(Wilks’ Lambda¼ 0.291; p , 0.01) and betweenmale and female subjects (Wilks’ Lambda¼ 0.552;p , 0.05). No interaction was found between groupaffiliation and gender. Follow-up ANOVA revealedsignificant differences between the groups for thevariable perfectionism (F(2,58)¼ 3.28; p , 0.05).Patients with FD attained the highest scores(24.3 6 4.8) in perfectionism compared with controlswho reached 21.9 6 3.3 (p , 0.05; contrast analysis)on a scale ranging from minimum ¼5 to maximum¼30. Figure 3 shows the findings of the perfectionismscale in the different groups.

Patients suffering from CP syndromes achievedscores (23.4 6 4.0) tendentially in between thehealthy musicians and those with FD. Differences inboth directions (CP vs. FD; CP vs. control) werenonsignificant.

Patients were asked to specify whether their atti-tudes concerning perfectionism were the same ordifferent before and after the onset of their disorders.All patients with FD and 19 out of 20 with CP wereable to give information about their attitudes towardperfectionism before onset of the disorders. All 20patients with FD (100%) estimated their attitudesconcerning perfectionism as the same before andafter onset. In one patient with CP, perfectionist



tendencies had increased after onset. Eighteen CPpatients (94.7%) had experienced no change in thisparameter. No difference was seen between perfec-tionistic tendencies of male and female subjects.Cronbach’s alpha was 0.72, displaying an adequateinternal consistency of the scale.

The QCC did not reveal any statistically significantdifferences between the groups (FD, CP, control) norbetween male and female subjects. In the FPI(Figure 4), follow-up ANOVA revealed significant dif-ferences between the groups for the subscales somaticcomplaints (F(2,58)¼ 10.16; p , 0.001) and emotionality(F(2,58)¼ 3.55; p , 0.05). Both patient groups showedmore somatic complaints than healthy musicians(FD: p¼ 0.01; CP: p , 0.001). Musicians sufferingfrom CP reached higher scores in the subscale of emo-tionality compared with healthy musicians (p , 0.05).

Gender differences were observed in two subscalesof the FPI-R: in the social orientation subscale, malemusicians had higher scores (6.3 6 1.4) than femalemusicians, who reached 5.1 6 1.5 (p , 0.01,F(1,58)¼ 11.07). Similarly, male musicians had moresomatic complaints (5.0 6 1.8) than female musicianswho had 4.0 6 1.8 (p¼ 0.01, F(1,58)¼ 6.9).

PERFECTIONISM AND ANXIETY INMUSICIAN’S DYSTONIA:PREEXISTENT OR PSYCHOREACTIVE?

The results demonstrated that psychological con-ditions in musicians with FD differed from those ofhealthy musicians. Anxiety disorders, and above all,social phobias, and specific phobias occurred moreoften in musicians with dystonia. Additionally, mu-sicians with FD were found to have highly perfec-tionist tendencies. Musicians with CP also showeddifferent psychological patterns from those of healthymusicians. They also more often reported anxiety,above all, free-floating anxiety. In the perfectionismscale, they recorded scores tendentially in betweenthose of the control group and the musicians withdystonia. In the FPI-R, both patient groups showedmore somatic complaints than healthy musicians.Furthermore, musicians with CP turned out to bemore emotional than controls. Taken together, acertain overlap was observed between the psycho-logical conditions of musicians with FD and thosesuffering from CP. However, musicians with dystoniashowed a pattern of exaggerated perfectionism, so-cial phobia, and specific phobias that was not seen inhealthy musicians or those with CP.

The question arises whether anxiety was presentbefore the playing-related disorders in both patientgroups and whether perfectionism was present inmusicians before they developed FD. Alternatively,both psychological features might reflect psychoreac-tive symptoms as a consequence of playing-related

� 26 December 2008 � 8:29 pm

FIGURE 3. Findings of the perfectionism scale of controls and musicians with chronic pain and those with focal dystonia.Left side: frequency distribution: percentage of subjects (of respective groups) who reached different perfectionism levels.Right side: results of all groups in the perfectionism scale: small boxes: mean values/large boxes: standard errors/whiskers:standard deviations (*p , 0.05).

ARTICLE IN PRESS

685686687688689690691692693694695696697698699700701702703704705706707708709710711712713714715716717718719720721722723724725726727728729730731732733734735736737738739740741742

743744745746747748749750751752753754755756757758759760761762763764765766767768769770771772773774775776777778779780781782

disorders. We hypothesized that these psychologicalconditions were already present in musicians beforethe onset of playing-related disorders. A prospectivestudy with the particular aim of assessing psycholog-ical conditions in musicians before the developmentof FD was not realizable due to a prevalence of FDbetween 1:200 and 1:500 in professional musicianswith the consequence of a low incidence.1 A retrospec-tive analysis was the only realizable approach. A ret-rospective inquiry was undertaken based onpersonal recollection. Under the given circumstances,this procedure was the only practical approach to ob-tain the desired information. As a limitation of thestudy, such retrospective reports are subject to biasand not always reliable. However, in view of the un-ambiguity and consistency of the reports of all pa-tients with dystonia, we postulate that perfectionismand anxiety have been preexistent and were not psy-choreactive phenomena.

In view of the findings for musicians with FD, thequestion arises whether and in which way thedescribed psychological conditions with anxietyand exaggerated perfectionism might be involvedin the etiology of FD in musicians.

FIGURE 4. Results of the FPI-R subscales: means of controls atonia. Stanine test values scaled from one (minimally pronouncedcontrol; **p , 0.01 compared to control).

783


THE POSSIBLE ROLE OFPERFECTIONISM AND ANXIETYDURING ONSET OF MUSICIAN’SDYSTONIA

Phenomenologically, musician’s dystonias are dys-functional movement patterns almost inextinguish-ably fixed in the procedural motor memory. How canthe observed psychological characteristics in musi-cians with dystonia be interpreted in context with theneurobiological basis of memory systems?

We suggest that specific affective conditions ofmaking music underlie the interaction between psy-chological phenomena and motor memory. Musicperformance is strongly linked to the emotions in away that is not comparable to any other activity inhuman life. On one hand, music is the ‘‘language ofemotions’’ and is able to communicate positive ornegative feelings. On the other hand, instrumentalmusic is based on extreme spatiotemporal sensori-motor precision, which can be scrutinized by both themusician and the audience. Therefore, many musi-cians experience strong and contrary feelings, withthe joy of performing alongside the fear of playing

nd musicians with chronic pain and those with focal dys-) to nine (maximally pronounced). (*p , 0.05 compared to

jej 2008 7

784785786787788789790791792793794795796797798799800

� 26 December 2008 � 8:29 pm

ARTICLE IN PRESS

801802803804805806807808809810811812813814815816817818819820821822823824825826827828829830831832833834835836837838839840841842843844845846847848849850851852853854855856857858

859860861862863864865866867868869870871872873874875876877878879880881882883884885886887888889890891892893894895896897898899900901902903904905906907908

wrong notes or the fear of failure, which reflects theextremely strict system of reward and punishment inprofessional musicianship. This double link to theemotions is a unique characteristic of makingmusic.51

We suggest that during onset of musician’s dysto-nia, this link between music performance and strongemotions may play a role in the establishment ofdystonic movement patterns in patients with perfec-tionism and anxiety. It is possible that emotionallyinduced motor memory consolidation may facilitatethe onset of dystonia in the subgroup of patients withthese psychological conditions. The process mightstart with the occurrence of a dystonic movement forwhich the cause is unknown. Musicians with aninclination toward anxiety and extreme perfection-ism may emphasize the disturbing and threateningelement in the occurred wrong movement. Thispsychological stress might induce a cascade of emo-tionally induced memory consolidation, which haspreviously been described and applied to differentforms of memory and which mainly relies on norad-renergic activation of the basolateral amygdala(BLA).52 The primary motor cortex, which is anessential locus of representation of digital motorsequences, receives a BLA projection.53 Thus, it maybe assumed that consolidation of early dystonicmovements as dysfunctional motor programs maybe facilitated by a BLA-mediated process in the pri-mary motor cortex.

Undoubtedly, the above outlined scenario consti-tutes only an epiphenomenon in a subgroup ofpatients with the described psychological conditions.Further support for a participation of limbic circuitsin the development of FD comes from the phenom-enologies of related FDs, for example, in calligra-phists, telegraphers, money counters, golfers etc. Twocommon features can be observed: 1) the necessity forhigh precision and 2) a strong emotional component.The first is related to the nature of the respectiveactivities, the latter originates in the fact that a strictsystem of reward and punishment underlies theseactivities. In sports, this is provided by victory anddefeat with the resulting advantages and disadvan-tages; the other activities are performed in profes-sional contexts, which means that people’s incomedepends on the quality of their work. One canspeculate that in all of these activities, an inclinationtoward anxiety and extreme perfectionism may alsofoster consolidation of dystonic movement patterns.

909910911912913914915

PARALLELS BETWEEN MUSICIANSWITH FOCAL DYSTONIA AND THOSEWITH CHRONIC PAIN

The findings in musicians with CP harmonize withother reports on CP patients. The association between



anxiety and CP has often been described for CPsyndromes in different sites (for example Ref. 54). Inparticular, social phobia has been found to be relatedto physically unexplained CP in a study with 130 pa-tients. Additionally, in keeping with the findings ofthe Hannover study, agoraphobia was found mini-mally pronounced in CP patients.55 Free-floating anx-iety, which was more often present in musicians withCP of this study, turned out to be a predictor for earlyretirement in patients with CP.56

The emotionality subscale (FPI-R), in which CPpatients had higher scores than the other groups,recorded substantial components of neuroticism,which was found to be more pronounced in patientswith CP in other studies.57

Finally, the FPI-R showed more somatic complaintsand higher levels of health concern in both patientgroups than in normal controls. It should be men-tioned that the questions for the somatic complaintssubscale did not focus on the diseases of the patientsspecifically, neither on FD, nor on CP syndromes. Thesimilarity in the findings of elevated somatic com-plaint scores and health concern scores of the FPI-R inboth patient groups might be based on a generallymore intense perception of somatic sensations indiseased musicians and a more health-focused ori-entation as an unspecific reaction to the disorders.

A certain overlap was seen between the psycho-logical conditions in musicians with FD and thosesuffering from CP. In some of the musicians withdystonia, intensified sensory input, as in CP syn-dromes, trauma, or nerve entrapment, precededFD.21 This CPeFD sequence means that some of themusicians with dystonia suffered from CP beforethey developed FD. That implies a certain overlapof both patient groups. However, among the sampleof musicians with dystonia of the Hannover study,CP had preceded FD in only one patient. Thus, theCPeFDesequence cannot explain the overlap in psy-chological findings of both groups.

The amount of perfectionism in musicians with CPwas ranked in between that in the control group andthe group with FD. The combination of a tendencytoward perfectionism and anxiety—either social pho-bia or free-floating anxiety—might promote a certainworking behavior, which might be a triggering factorfor both disorders, FD and CP syndromes.

CONCLUSION AND CONSEQUENCES

In summary, special psychological conditions in-cluding anxiety and extreme perfectionism were seenin musicians with dystonia. We suggest that theseconditions may facilitate the onset of the disorder. Itshould be emphasized that the emotional aspects ofmusic performance, specifically the enormous pro-fessional pressure, substantially contribute to stress-

916

� 26 December 2008 � 8:29 pm

ARTICLE IN PRESS

917918919920921922923924925926927928929930931932933934935936937938939940941942943944945946947948949950951952953954955956957958959960961962963964965966967968969970971972973974

975976977978979980981982983984985986987988989990991992993994995996997998999

1000100110021003100410051006

induced processes that may foster consolidation ofdystonic movements. In part, the unyielding rewardand punishment frame in the reproductive classicalmusic scene provides a fertile ground for thesestresses in musicians. This in turn could explainwhy, for example, improvising Jazz musicians aremuch less likely to develop musician’s dystonia.Here as in many other music cultures, reproductionof the precise musical notation plays only a minorrole. Learning is frequently based on imitation andmovements frequently can be selected deliberately,obeying the individual’s anatomical prerequisites.

In Figure 5 we propose a model, specifying the roleof anxiety and perfectionism in triggering musician’sdystonia. Furthermore, the possible co-action be-tween predisposition and intrinsic and extrinsic trig-gering factors is displayed.

With this model, we now have means at handcontributing to the prevention of musician’s dysto-nia. Preventing dystonia is important, since success-ful treatment is still a challenge. Many of the availablemedical approaches are only moderately effective,and other options have yet to be developed.Behavioral therapies and interdisciplinary strategiescombining pharmacological and pedagogicalmethods are promising, but the different approachesneed to be evaluated, and long-term effects are stillunknown (for a review see Ref. 58).

Concerning prevention, exaggerated perfectionismand anxiety as triggering factors should be addressedin the education of musicians. This has to be started atearly infancy. From the first lesson on, music

FIGURE 5. The possible co-action between predisposition and inof musician’s dystonia. Further explanations are given in the te


educators should strive to create a friendly, support-ive atmosphere focusing on creativity, curiosity, andplayful experiences in the world of sounds. It is notby chance that we commonly speak of ‘‘playing aninstrument’’ and not of ‘‘working an instrument.’’ Ofcourse, structured, goal-directed learning is a pre-requisite of musical mastery. Here, reasonable prac-tice schedules, economic technique, prevention ofoveruse and pain, mental practice, variations ofmovement patterns, maintenance of motivation andavoidance of mechanical repetitions and frustration,healthy living habits, warm-ups and cool-downexercises, regular physical exercise, sufficient breaks,and sleep are the cornerstones of healthy musicalpractice.

Finally, the role of societal constraints should notbe neglected. In the last decades, the classical musicsector was inundated by CD recordings of peers inthe fields. Frequently, these recordings can be re-garded as ‘‘laboratory music,’’ composed in thetranquility of the studios with the help of toneengineers and electronic ‘‘re-mastering.’’ These re-cordings are considered as the ‘‘gold standard.’’ Theyfrequently create unrealistic expectations in listenersand interpreters, adding stress to the performers. Inaddition, our classical music culture reflects thegeneral societal pressures of the developed countries.Highest precision and efficiency are the demands weall are subjected to. In music, this frequently createsan attitude of great artistic accomplishment, which,however, frequently is not nurtured by a sufficientpersonal expression of emotional experience. The

trinsic and extrinsic triggering factors in the manifestationxt.

jej 2008 9

10071008100910101011101210131014101510161017101810191020102110221023102410251026102710281029103010311032

� 26 December 2008 � 8:29 pm

½Q3�

ARTICLE IN PRESS

1033103410351036103710381039104010411042104310441045104610471048104910501051105210531054105510561057105810591060106110621063106410651066106710681069107010711072107310741075107610771078107910801081108210831084108510861087108810891090

1091109210931094109510961097

latter, of course, has to be collected somewhereoutside the practicing room. As a consequence, we,therefore, should correct our expectations and listen-ing habits, replacing the fascination of mere perfec-tion and virtuosity by the joy of emotionalcommunication shared with the audience and themusicians.

109810991100110111021103110411051106110711081109111011111112111311141115111611171118111911201121112211231124112511261127112811291130113111321133113411351136113711381139114011411142114311441145114611471148

REFERENCE

1. Altenmuller E. Focal dystonia: advances in brain imaging andunderstanding of fine motor control in musicians. Hand Clin.2003;19:523–38.

2. Nutt JG, Muenter MD, Melton LJ, Aronson A, Kurland LT.Epidemiology of dystonia in Rochester, Minnesota. AdvNeurol. 1988;50:361–5.

3. Nakashima K, Kusumi M, Inoue Y, Takahashi K. Prevalence offocal dystonias in the western area of Tottori Prefecture inJapan. Mov Disord. 1995;10:440–3.

4. Schmidt A, Jabusch HC, Altenmuller E, et al. Dominantlytransmitted focal dystonia in families of patients with musi-cians cramp. Neurology. 2006;67:691–3.

5. Naumann M, Pirker W, Reiners K, Lange KW, Becker G, BruckeT. Imaging the pre- and postsynaptic side of striatal dopamin-ergic synapses in idiopathic cervical dystonia: a SPECT studyusing [123I] epidepride and [123I] beta-CIT. Mov Disord.1998;13:319–23.

6. Preibisch C, Berg D, Hofmann E, Solymosi L, Naumann M. Ce-rebral activation patterns in patients with writer’s cramp: afunctional magnetic resonance imaging study. J Neurol. 2001;248:10–7.

7. Lenz FA, Byl NN. Reorganization in the cutaneous core of thehuman thalamic principal somatic sensory nucleus (Ventralcaudal) in patients with dystonia. J Neurophysiol. 1999;82:3204–12.

8. Delmaire C, Krainik A, Tezenas du Montcel S, et al. Disorga-nized somatotopy in the putamen of patients with focal handdystonia. Neurology. 2005;64:1391–6.

9. Byl NN, Merzenich MM, Jenkins WM. A primate genesismodel of focal dystonia and repetitive strain injury: I. Learn-ing-induced dedifferentiation of the representation of thehand in the primary somatosensory cortex in adult monkeys.Neurology. 1996;47:508–20.

10. Bara-Jiminez W, Catalan MJ, Hallett M, Gerloff C. Abnormalsensory homunculus in dystonia of the hand. Ann Neurol.1998;44:828–31.

11. McKenzie AL, Nagarajan SS, Roberts TP, Merzenich MM, BylNN. Somatosensory representation of the digits and clinicalperformance in patients with focal hand dystonia. Am J PhysMed Rehabil. 2003;82:737–49.

12. Elbert T, Candia V, Altenmuller E, et al. Alteration of digitalrepresentations in somatosensory cortex in focal hand dysto-nia. NeuroReport. 1998;16:3571–5.

13. Elbert T, Pantev C, Wienbruch C, Rockstroh B, Taub E. In-creased cortical representation of the fingers of the left handin string players. Science. 1995;270:305–7.

14. Altenmuller E. From the Neanderthal to the concert hall: devel-opment of sensory motor skills and brain plasticity in musicperformance. In: Williamon A, Coimbra D (eds). Proceedingsof the international symposium on performance science.Utrecht: European Association of Conservatoires (AEC),2007:1–14.

15. Ragert P, Schmidt A, Altenmuller E, Dinse R. Meta-plasticity inmusicians. Eur J Neurosci. 2004;19:473–8.

16. Naumann M, Magyar-Lehmann S, Reiners K, Erbguth F,Leenders KL. Sensory tricks in cervical dystonia: perceptualdysbalance of parietal cortex modulates frontal motor pro-gramming. Ann Neurol. 2000;47:322–8.

17. Jabusch HC, Altenmuller E. Epidemiology, phenomenologyand therapy of musician’s cramp. In: Altenmuller E, Kesselring



J, Wiesendanger M (eds). Music, motor control and the brain.Oxford: Oxford University Press, 2006:265–82.

18. Lederman RJ. Focal dystonia in instrumentalists: clinical fea-tures. Med Probl Perform Art. 1991;6:132–6.

19. Lim V, Altenmuller E. Musicians cramp: instrumental and gen-der differences. Med Probl Perform Art. 2003;18:21–7.

20. Schmidt A, Jabusch HC, Altenmuller E, et al. Clinical geneticsof musician’s dystonia: familial aggregation of dystonia andother movement disorders. Neurology, in press.

21. Charness ME, Ross MH, Shefner JM. Ulnar neuropathy anddystonic flexion of the fourth and fifth digits: clinical correla-tion in musicians. Muscle Nerve. 1996;19:431–7.

22. Chamagne P. Functional dystonia in musicians: rehabilitation.Hand Clin. 2003;19:309–16.

23. Brandfonbrener AG, Robson C. A review of 111 musicians withfocal dystonia seen at a performing artist’s clinic 1985e2002.Mov Disord. 2002;17:1135–8.

24. Schuele S, Lederman RJ. Focal dystonia in woodwind instru-mentalists: long-term outcome. Med Probl Perform Art. 2003;18:15–20.

25. Gowers WR. A manual of diseases of the nervous system. Lon-don: Churchill, 1888.

26. Brain WR. Diseases of the nervous system. London: OxfordUniversity Press, 1933.

27. Sheehy MP, Marsden CD. Writer’s cramp e a focal dystonia.Brain. 1982;105:461–80.

28. Marsden CD, Sheehy MP. Writer’s cramp [Review]TrendsNeurosci. 1990;13:148–53.

29. Frommer J. Idiopathic writer’s cramp as a psychosomatic dis-order. Z Psychosom Med. 1992;38:49–62.

30. Schulze A, Jacob HW, Guckler A. Psychosomatic determinantsof writer’s cramp. Psychother Psychosom Med Psychol. 1992;42:201–5.

31. Windgassen K, Ludolph A. Psychiatric aspects of writer’scramp. Eur Arch Psychiatry Clin Neurosci. 1991;241:170–6.

32. Pai MN. The nature and treatment of writer’s cramp. J MentSci. 1947;93:68–81.

33. Gibson HB. Writer’s cramp: a behavioural approach. BehavRes Ther. 1972;10:371–80.

34. Cottraux JA, Juenet C, Collet L. The treatment of writer’scramp with multimodal behaviour therapy and biofeedback:a study of 15 cases. Br J Psychiatry. 1983;142:180–3.

35. Zacher A. Writer’s cramp e focal dystonia or psychogenicmovement disorder? A critical literature study [Review]-Fortschr Neurol Psychat. 1989;57:328–36.

36. Ludolph AC, Windgassen K. Writer’s cramp: a study of 30cases. Nervenarzt. 1992;63:462–6.

37. Harrington RC, Wieck A, Marks IM, Marsden CD. Writer’scramp: not associated with anxiety. Mov Disord. 1988;3:195–200.

38. Gundel H, Busch R, Ceballos-Baumann AA, Seifert E. Psychi-atric comorbidity in patients with spasmodic dysphonia: a con-trolled study. J Neurol Neurosurg Psychiatry. 2008;78:1398–400.

39. Bugalho P, Correa B, Guimaraes J, Xavier M. Set-shifting andbehavioral dysfunction in primary focal dystonia. Mov Disord.2008;23:200–6.

40. Herrojo-Ruiz M, Senghaas P, Grossbach M, et al. Defective in-hibition and inter-regional phase synchronization in pianistswith musician’s dystonia (MD): an EEG study. Hum BrainMapp, in press.

41. Rosenkranz K, Williamon A, Butler K, Cordivari C, LeesAJ, Rothwell JC. Pathophysiological differences betweenmusicians dystonia and writers cramp. Brain. 2005;128:918–31.

42. Altenmuller E. Robert Schumann’s focal dystonia. Bogous-slavsky E, , Boller E (eds). Neurological disorders on fa-mous artists. Front Neurol Neurosci. 2005:179–88, Karger,Basel.

43. Altenmuller E. The end of the song: Robert Schumann’sfocal dystonia. In: Altenmuller E, Kesselring J, WiesendangerM (eds). Music, motor control and the brain. Oxford: OxfordUniversity Press, 2006:251–64.

� 26 December 2008 � 8:29 pm

ARTICLE IN PRESS

1149

1150

1151

1152

1153

1154

1155

1156

1157

1158

1159

1160

1161

1162

1163

1164

1165

1166

1167

1168

1169

1170

1171

1172

1173

1174

1175

1176

1177

1178

1179

1180

1181

1182

1183

1184

1185

1186

1187

1188

1189

1190

1191

1192

44. Jabusch HC, Muller SV, Altenmuller E. Anxiety in musicianswith focal dystonia and those with chronic pain. Mov Disord.2004;19:1169–75.

45. Jabusch HC, Altenmuller E. Anxiety as an aggravating factorduring onset of focal dystonia in musicians. Med Probl Per-form Art. 2004;192:75–81.

46. Newmark J, Hochberg FH. Isolated painless manual incoordi-nation in 57 musicians. J Neurol Neurosurg Psychiatry. 1987;503:291–5.

47. Smith BH, Elliott AM, Chambers WA, Smith WC, HannafordPC, Penny K. The impact of chronic pain in the community.Fam Pract. 2001;18:292–9.

48. Fishbein M, Middlestadt E, Ottati V, Straus S, Ellis A. Medicalproblems among ICSOM musicians: overview of a nationalsurvey. Med Probl Perform Art. 1988;3:1–8.

49. Fahrenberg J, Hampel R, Selg H. Das Freiburger Persoenlich-keitsinventar FPI. Revidierte Fassung FPI-R und teilweisegeaenderte Fassung FPI-A1. Goettingen: Hogrefe-Verlag fuerPsychologie, 1994.

50. Krampen G. Fragebogen zu Kompetenz- und Kontrollueber-zeugungen (FKK). Goettingen: Hogrefe-Verlag fuer Psycholo-gie, 1991.


51. Altenmuller E. Causes et traitements de la dystonie de fonctionchez les musiciens. Medicine des arts. 2001;36:19–27.

52. McGaugh JL. Memory e a century of consolidation [Review]S-cience. 2000;287:248–51.

53. Sripanidkulchai K, Sripanidkulchai B, Wyss JM. The corticalprojection of the basolateral amygdaloid nucleus in the rat: aretrograde fluorescent dye study. J Comp Neurol. 1984;229:419–31.

54. Craig KD. Emotional aspects of pain. In: Wall PD, Melzack R(eds). Textbook of pain. Edinburgh: Churchill Livingstone,1984:153–61.

55. Linton SJ. A review of psychological risk factors in back andneck pain [Review]Spine. 2000;25:1148–56.

56. Haerkaepaeae K. Psychosocial factors as predictors for earlyretirement in patients with chronic low back pain. J PsychosomRes. 1992;36:553–9.

57. Harkins SW, Price DD, Braith J. Effects of extraversion andneuroticism on experimental pain, clinical pain, and illness be-havior. Pain. 1989;36:209–18.

58. Jabusch HC, Altenmuller E. Focal Dystonia in musicians:from phenomenology to therapy. Adv Cogn Psychol. 2006;2:207–20.

jej 2008 11

1193

1194

� 26 December 2008 � 8:29 pm

Focal hand dystonia in musicians: phenomenology, etiology, and psychological trigger factors

Documents

Transcript of Focal hand dystonia in musicians: phenomenology, etiology, and psychological trigger factors