Weaver’s dystonia – a novel form of focal task specific dystonia Authors A. Narendrakumar Barad Consultant Neurologist & Movement disorders specialist Sterling Hospital, Ahmedabad, Gujarat, India 380052 +919099075332 Email id – [email protected]

Running title – “Weaver’s dystonia – another form of focal task specific dystonia” Keywords – weaver’s dystonia, task specific dystonia, occupational dystonia, writer’s cramp

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Task-specific dystonias (TSDs) are idiopathic focal dystonias characterised by abnormal posturing of a body part during selective motor tasks that often involve highly skilled and repetitive movements of muscles.1,2 For much of the 20th century, these TSDs were considered psychogenic and given the name of occupational neuroses. In 1982, Sheehy and Marsden described the real dystonic features and lack of psychopathology in their series of patients with task-specific dystonias.3 Upper extremity and lower facial muscles are commonly involved in different TSDs. Described upper extremity TSDs related to profession includes shoemaker’s dystonia, tailor’s dystonia, pianist’s dystonia, writer’s cramps, hairdresser and telegraphist’s cramps, while some upper limb TSDs related to sports includes the pistol shooter’s cramp,4 the golfer’s yips,5 and petanque player’s arm dystonia.6 Rarely, TSDs also affect the cervical region and lower limbs.7,8 Hereby, I am describing a novel form of TSD related to weaving profession- weavers dystonia. Weaving is an old traditional method of making clothes and other crafts in certain parts of India, in which they interlace yarns with fine and repetitive movements of the hands to make colourful weaven cloths or showpieces. Basic weaving includes two types of yarns/threads, one longitudinal called warp and a lateral one called weft. Weaving task requires highly skilled repetition of certain hand movements for perfect angled weaving of the threads to produce a plain, decorative or artistic design. Though, most of the companies nowadays use automatic weaving machine, some low budget companies in India are still employing human workers for weaving work. Case report This 34-year-old, right-handed man has been working in a weaving company for the last 24 years. He presented to me with the complain of abnormal posturing of left hand and forearm, during weaving work for the last 20 years. This complaint was insidious in onset and slowly progressive in nature. After 10 years of symptoms onset, he had extreme difficulty in doing this weaving work and so finally he stopped the work. He had no difficulty in using left hand for other activities. He tried another job which involved clerical work for 5 years, but he was not able to sustain his family financially. So, he returned to his previous weaving job. He found no difficulty in doing the same weaving work for the first 5 years after restarting, but subsequently, he redeveloped the same symptoms in the same hand. He denied previous trauma or any exposure to neuroleptics or toxins. There was no family history of consanguinity, dystonia or other neurological problems. His birth history and developmental history were unremarkable. Upon my consultation, the clinical and neurological examination was unremarkable except abnormal flexion posturing of the left hand, adduction and flexion of first four fingers with extension of little finger, ulnar deviation of wrist, loss of dexterity and slowness in executing the particular movement of putting yarns in a hook during his factory work (video-1 and 2). His routine blood investigations (Complete blood count, Renal & Liver function tests, Serum

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Electrolytes, Thyroid and Lipid profile) serum copper and ceruloplasmin levels and imaging of Central nervous system (Mri brain with SWI and CT scan of brain) were normal. Discussion TSDs are a form of isolated focal dystonias with a peculiar feature of its occurrence only during the performance of a highly skilled motor task at least initially.2 Multiple theories on the pathophysiology of task-specific dystonias have been described.9 Recently, the role of cerebellum in the form of aberrant afferent inputs to basal ganglia is also highlighted.10 Our patient demonstrated a task specific wrist flexion and fingers adduction with flexion dystonia after many years of fine and repetitive hand movements for weaving. Indeed, he had no difficulty in using the same hand for other activities like buttoning/unbuttoning and eating. So this entity can be considered under the spectrum of TSDs. Clinical course and unique characteristics of TSDs make consideration of other differentials very unlikely for the given case. Many forms of TSDs are described in different professions; however, it has not been described in relation to weaving work, which makes it a novel form of TSDs. In addition, recurrence of same symptoms even after 5 years of interruption of particular task suggests possibility of underlying dynamic basal ganglia network abnormality. Such an aberrant network can be deactivated in a given individual on stopping that particular task due to possible sensorimotor retuning.11 However, as per experience-based medicine in most of the cases and also as in our case, these networks can be reactivated on exposure to the same highly skilled repetitive task in a given individual. However, further scientific research to support this clinical hypothesis of “dynamic aberrant basal ganglia network for TSDs” is needed. References 1.

Albanese, A. et al. Phenomenology and classification of dystonia: A consensus update. Mov. Disord. 28, 863–873 (2013).

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Torres-Russotto, D. & Perlmutter, J. S. Task-specific dystonias: A review. Annals of the New York Academy of Sciences 1142, 179–199 (2008).

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Writers’ cramp-a focal dystonia. - PubMed - NCBI. Available at: https://www.ncbi.nlm.nih.gov/pubmed/7104663. (Accessed: 2nd May 2020)

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Sitburana, O. & Ondo, W. G. Task-specific focal hand dystonia in a professional pistolshooter. Clinical Neurology and Neurosurgery 110, 423–424 (2008).

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Smith, A. M. et al. A multidisciplinary study of the ‘Yips’ phenomenon in golf: An exploratory analysis. Sport. Med. 30, 423–437 (2000).

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Lagueny, A. et al. Freezing of shoulder flexion impeding boule throwing: A form of

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task-specific focal dystonia in petanque players. Mov. Disord. 17, 1092–1095 (2002). 7.

Ramdhani, R. A. & Frucht, S. J. Adult-onset Idiopathic Focal Lower Extremity Dystonia: A Rare Task-Specific Dystonia. Tremor Other Hyperkinet. Mov. (N. Y). 3, (2013).

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Kaji, R., Bhatia, K. & Graybiel, A. M. Pathogenesis of dystonia: is it of cerebellar or basal ganglia origin? Journal of neurology, neurosurgery, and psychiatry 89, 488–492 (2018).

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Candia, V. et al. Sensory motor retuning: A behavioral treatment for focal hand dystonia of pianists and guitarists. Arch. Phys. Med. Rehabil. 83, 1342–1348 (2002).

Disclosures 1) Funding sources and conflict of interest: No specific funding was received for this work. The author declares that there are no conflicts of interest relevant to this work. 2) Financial disclosures for the previous 12 months: The author declares that there are no additional disclosures to report. Ethical Compliance Statement Written informed consent for video and case publication was obtained. We confirm that we have read the journal’s position on issues involved in ethical publication and affirm that this work is consistent with those guidelines. The author is very much aware about his patient’s work profile and the issue of child labour; however, the author is not supporting or favouring child labour by any means. Author roles1. Research project: A. Conception, B. Organization, C. Execution; 2. Statistical Analysis: A. Design, B. Execution, C. Review and Critique; 3. Manuscript Preparation: A. Writing of the first draft, B. Review and Critique; NB: 3A, 3B Legends to the video Videos 1 & 2 – patient is doing weaving task at his company, whenever he uses his left hand for taking the thread from one side and tries to put it in a hook near his body, you can see the

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difficulty in holding and hooking the thread along with some flexion posturing of left hand, ulnar deviation of the wrist, and adduction of first four fingers with little finger extension.

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Weaver’s dystonia – a novel form of focal task specific dystonia Authors A. Narendrakumar Barad Consultant Neurologist & Movement disorders specialist...
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