Fixel‐Based Analysis Reveals Whole‐Brain White Matter Abnormalities in Cervical Dystonia

Author:

Zito Giuseppe A.1ORCID,Tarrano Clément23ORCID,Ouarab Salim2,Jegatheesan Prasanthi2,Ekmen Asya2,Béranger Benoît4,Valabregue Romain4,Hubsch Cécile2,Sangla Sophie2,Bonnet Cécilia2,Delorme Cécile2,Méneret Aurélie25ORCID,Degos Bertrand267ORCID,Bouquet Floriane2,Apoil Brissard Marion8,Vidailhet Marie25ORCID,Hasboun Dominique23,Worbe Yulia29,Roze Emmanuel25,Gallea Cécile2ORCID

Affiliation:

1. Swiss Paraplegic Research Nottwil Switzerland

2. Movement Investigation and Therapeutics Team, Paris Brain Institute, Sorbonne University, Inserm U1127, CNRS UMR7225 Paris France

3. Department of Neurology Assistance Publique‐Hôpitaux de Paris, Pitié‐Salpêtrière Hospital Paris France

4. Center for NeuroImaging Research (CENIR) Paris Brain Institute, Sorbonne University, Inserm U1127, CNRS UMR 7225 Paris France

5. DMU Neurosciences, Assistance Publique‐Hôpitaux de Paris Paris France

6. Neurology Unit AP‐HP, Avicenne University Hospital, Sorbonne Paris Nord Bobigny France

7. Center for Interdisciplinary Research in Biology, Collège de France CNRS UMR7241/INSERM U1050, Université PSL Paris France

8. Université de Caen Normandie Caen France

9. Department of Neurophysiology Saint‐Antoine Hospital, Assistance Publique‐Hôpitaux de Paris Paris France

Abstract

AbstractBackgroundCervical dystonia (CD) is a form of isolated focal dystonia typically associated to abnormal head, neck, and shoulder movements and postures. The complexity of the clinical presentation limits the investigation of its pathophysiological mechanisms, and the neural networks associated to specific motor manifestations are still the object of debate.ObjectivesWe investigated the morphometric properties of white matter fibers in CD and explored the networks associated with motor symptoms, while regressing out nonmotor scores.MethodsNineteen patients affected by CD and 21 healthy controls underwent diffusion‐weighted magnetic resonance imaging. We performed fixel‐based analysis, a novel method evaluating fiber orientation within specific fiber bundles, and compared fiber morphometric properties between groups. Moreover, we correlated fiber morphometry with the severity of motor symptoms in patients.ResultsCompared to controls, patients exhibited decreased white matter fibers in the right striatum. Motor symptom severity negatively correlated with white matter fibers passing through inferior parietal areas and the head representation area of the motor cortex.ConclusionsAbnormal white matter integrity at the basal ganglia level may affect several functional networks involved, for instance, in motor preparation and execution, visuomotor coordination, and multimodal integration. This may result in progressive maladaptive plasticity, culminating in overt symptoms of dystonia. © 2023 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.

Funder

Agence Nationale de la Recherche

Amadys

Schweizerischer Nationalfonds zur Förderung der Wissenschaftlichen Forschung

Fondation de France

Medtronic

Boston Scientific

Ipsen Biopharmaceuticals

Allergan

Fondation Desmarest

Merck

Dystonia Medical Research Foundation

European Academy of Neurology

Publisher

Wiley

Subject

Neurology (clinical),Neurology

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