@article{da3c7f1027374c43b0ce8c91d852b39e,
title = "Involuntary thumb flexion on neurological examination: An unusual form of upper limb dystonia in the faroe islands",
abstract = "Background: The prevalence of dystonia varies worldwide. A prior report suggested a high prevalence of focal dystonia in the Faroese population, possibly reflecting a founder effect. During standardized neurological examination as part of an ongoing neuroepidemiologic study in the Faroe Islands, we noted an unusual phenomenon of thumb flexion during repetitive hand movements in a subset of subjects and sought to define its phenomenology. Methods: We requested commentary from a panel of dystonia experts regarding the phenomenology of the movements. These experts reviewed the videotaped neurological examination. Results: Among the experts, dystonia was the leading diagnosis. Alternate causes were considered, but deemed less likely. Discussion: Diagnosis of dystonia requires careful clinical assessment and consideration of associated features. We report a novel form of dystonia, not previously described to our knowledge, in this isolated population. Further studies of dystonia prevalence in the Faroe Islands are merited to characterize its burden in this population and its specific clinical characteristics.",
keywords = "Dystonia, Faroe Islands, Focal dystonia, Phenomenology, Thumb flexion",
author = "Kim, {Christine Y.} and Petersen, {Maria Skaalum} and Eliasen, {Eina H.} and Giovanni Defazio and Paul Greene and Jinnah, {Hyder A.} and Tijssen, {Marina A.J.} and Louis, {Elan D.}",
note = "Funding Information: *To whom correspondence should be addressed. E-mail:christine.kim@yale.edu Editor: Julian Benito-Leon, Hospital “12 de Octubre,” Spain Received: June 18, 2019; Accepted: July 23, 2019; Published: August 20, 2019 Copyright: {\textcopyright} 2019 Kim et al. This is an open-access article distributed under the terms of the Creative Commons Attribution–Noncommercial–No Derivatives License, which permits the user to copy, distribute, and transmit the work provided that the original authors and source are credited; that no commercial use is made of the work; and that the work is not altered or transformed. Funding: This work was supported by NIH grants R01 NS039422 and NS094607 (E.H.E., E.D.L., M.S.P.). Financial Disclosures: Dr. Jinnah reports grant support unrelated to the article from Cavion Therapeutics, Ipsen Pharmaceuticals, and Retrophin Inc. Dr. Jinnah has also served on advisory boards or as a consultant for Allergan Inc., CoA Therapeutics, Medtronic Inc., and Retrophin Inc. He is also the principal investigator for the Dystonia Coalition, which receives the majority of its support through NIH grant TR001456 from the Office of Rare Diseases Research at the National Center for Advancing Translational Sciences, and previously NS065701 from the National Institutes of Neurological Disorders and Stroke. The Dystonia Coalition has received additional material or administrative support from industry sponsors (Allergan Inc. and Merz Pharmaceuticals) as well as private foundations (The American Dystonia Society, Beat Dystonia, The Benign Essential Blepharospasm Foundation, Cure Dystonia Now, Dystonia Europe, Dystonia Inc., Dystonia Ireland, The Dystonia Medical Research Foundation, The Foundation for Dystonia Research, The National Spasmodic Dysphonia Association, and The National Spasmodic Torticollis Association). Conflicts of Interest: The authors report no conflicts of interest. Ethics statement: This study was performed in accordance with the ethical standards detailed in the Declaration of Helsinki. The authors{\textquoteright} institutional ethics committee has approved this study and all patients provided written informed consent. Publisher Copyright: {\textcopyright} 2019 Kim et al.",
year = "2019",
doi = "10.7916/tohm.v0.686",
language = "English (US)",
volume = "9",
pages = "1--7",
journal = "Tremor and Other Hyperkinetic Movements",
issn = "2160-8288",
}