Psychiatric phenotype in neurodevelopmental myoclonus-dystonia is underpinned by abnormality of cerebellar modulation on the cerebral cortex.


Journal

Scientific reports
ISSN: 2045-2322
Titre abrégé: Sci Rep
Pays: England
ID NLM: 101563288

Informations de publication

Date de publication:
27 Sep 2024
Historique:
received: 27 03 2024
accepted: 17 09 2024
medline: 28 9 2024
pubmed: 28 9 2024
entrez: 28 9 2024
Statut: epublish

Résumé

Psychiatric symptoms are common in neurodevelopmental movement disorders, including some types of dystonia. However, research has mainly focused on motor manifestations and underlying circuits. Myoclonus-dystonia is a rare and homogeneous neurodevelopmental condition serving as an illustrative paradigm of childhood-onset dystonias, associated with psychiatric symptoms. Here, we assessed the prevalence of psychiatric disorders and the severity of depressive symptoms in patients with myoclonus-dystonia and healthy volunteers (HV). Using resting-state functional neuroimaging, we compared the effective connectivity within and among non-motor and motor brain networks between patients and HV. We further explored the hierarchical organization of these networks and examined the relationship between their connectivity and the depressive symptoms. Comparing 19 patients to 25 HV, we found a higher prevalence of anxiety disorders and more depressive symptoms in the patient group. Patients exhibited abnormal modulation of the cerebellum on the cerebral cortex in the sensorimotor, dorsal attention, salience, and default mode networks. Moreover, the salience network activity was directed by the cerebellum in patients and was related to depressive symptoms. Altogether, our findings highlight the role of the cerebellar drive on both motor and non-motor cortical areas in this disorder, suggesting cerebellar involvement in the complex phenotype of such neurodevelopmental movement disorders.

Identifiants

pubmed: 39333780
doi: 10.1038/s41598-024-73386-9
pii: 10.1038/s41598-024-73386-9
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

22341

Subventions

Organisme : European Commission
ID : EJP RD COFUND-EJP N° 825575 - EurDyscover
Organisme : AMADYS
ID : Fonds de Dotation Brou de Laurière

Informations de copyright

© 2024. The Author(s).

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Auteurs

Clément Tarrano (C)

Paris Brain Institute, Sorbonne University - ICM, Inserm CNRS, Paris, F-75013, France.
Department of Neurology, Clinical Investigation Center for Neurosciences, Assistance Publique-Hôpitaux de Paris, Pitié-Salpêtrière Hospital, Paris, France.
Department of Neurophysiology, Saint-Antoine Hospital, Paris, France.

Cécile Galléa (C)

Paris Brain Institute, Sorbonne University - ICM, Inserm CNRS, Paris, F-75013, France.
Centre de NeuroImagerie de Recherche (CENIR), Sorbonne Université, UMR S 975, CNRS UMR 7225, ICM, Paris, F-75013, France.

Cécile Delorme (C)

Paris Brain Institute, Sorbonne University - ICM, Inserm CNRS, Paris, F-75013, France.
Department of Neurology, Clinical Investigation Center for Neurosciences, Assistance Publique-Hôpitaux de Paris, Pitié-Salpêtrière Hospital, Paris, France.

Eavan M McGovern (EM)

Department of Neurology, St Vincent's University Hospital Dublin, Dublin, Ireland.

Cyril Atkinson-Clement (C)

Paris Brain Institute, Sorbonne University - ICM, Inserm CNRS, Paris, F-75013, France.
School of Medicine, University of Nottingham, Nottingham, UK.

Vanessa Brochard (V)

Department of Neurology, Clinical Investigation Center for Neurosciences, Assistance Publique-Hôpitaux de Paris, Pitié-Salpêtrière Hospital, Paris, France.

Stéphane Thobois (S)

Department of Neurology, Hospices Civils de Lyon, Pierre Wertheimer Neurological Hospital, Expert Parkinson Center NS-PARK/FCRIN, Bron, France.
Marc Jeannerod Cognitive Neuroscience Institute, CNRS, UMR 5229, Bron, France.
University of Lyon, Université Claude Bernard Lyon 1, Faculté de Médecine et de Maïeutique Lyon Sud Charles Mérieux, Oullins, France.

Christine Tranchant (C)

Département of Neurology, Universitary Hospital of Strasbourg, Strasbourg, France.
Institut de Génétique et de Biologie Moléculaire et Cellulaire (IGBMC), INSERM- U964/CNRS-UMR7104/ University of Strasbourg, Illkirch, France.
Fédération de Médecine Translationnelle de Strasbourg (FMTS), University of Strasbourg, Strasbourg, France.

David Grabli (D)

Department of Neurology, Clinical Investigation Center for Neurosciences, Assistance Publique-Hôpitaux de Paris, Pitié-Salpêtrière Hospital, Paris, France.

Bertrand Degos (B)

Department of Neurology, Avicenne Hospital, Assistance Publique-Hôpitaux de Paris, Sorbonne Paris Nord, Bobigny, France.

Jean Christophe Corvol (JC)

Paris Brain Institute, Sorbonne University - ICM, Inserm CNRS, Paris, F-75013, France.
Department of Neurology, Clinical Investigation Center for Neurosciences, Assistance Publique-Hôpitaux de Paris, Pitié-Salpêtrière Hospital, Paris, France.

Jean-Michel Pedespan (JM)

Department of Neuropediatry, Universitary Hospital of Pellegrin, Bordeaux, France.

Pierre Krystkowiak (P)

, Abu Dhabi Stem Cells Centre, Abu Dhabi, United Arab Emirates.

Jean-Luc Houeto (JL)

Department of Neurology CHU Limoges, EpiMaCT - Epidemiology of chronic diseases in tropical zone, Institute of Epidemiology and Tropical Neurology, Inserm U1094, IRD U270, Univ. Limoges, OmegaHealth, Limoges, France.

Adrian Degardin (A)

Department of Neurology, Tourcoing Hospital, Tourcoing, France.

Luc Defebvre (L)

, Troubles cognitifs dégénératifs et vasculaires, Lille, F-59000, France.
Lille Centre of Excellence for Neurodegenerative Diseases (LiCEND), University of Lille, CHU Lille, INSERM, Lille, U1172, F-59000, France.

Benoit Beranger (B)

Paris Brain Institute, Sorbonne University - ICM, Inserm CNRS, Paris, F-75013, France.
Centre de NeuroImagerie de Recherche (CENIR), Sorbonne Université, UMR S 975, CNRS UMR 7225, ICM, Paris, F-75013, France.

Davide Martino (D)

Department of Clinical Neurosciences, Cumming School of Medicine, University of Calgary, Calgary, AB, Canada.
Hotchkiss Brain Institute, University of Calgary, Calgary, AB, Canada.

Emmanuelle Apartis (E)

Paris Brain Institute, Sorbonne University - ICM, Inserm CNRS, Paris, F-75013, France.
Department of Neurophysiology, Saint-Antoine Hospital, Paris, France.

Marie Vidailhet (M)

Paris Brain Institute, Sorbonne University - ICM, Inserm CNRS, Paris, F-75013, France.
Department of Neurology, Clinical Investigation Center for Neurosciences, Assistance Publique-Hôpitaux de Paris, Pitié-Salpêtrière Hospital, Paris, France.

Emmanuel Roze (E)

Paris Brain Institute, Sorbonne University - ICM, Inserm CNRS, Paris, F-75013, France.
Department of Neurology, Clinical Investigation Center for Neurosciences, Assistance Publique-Hôpitaux de Paris, Pitié-Salpêtrière Hospital, Paris, France.

Yulia Worbe (Y)

Paris Brain Institute, Sorbonne University - ICM, Inserm CNRS, Paris, F-75013, France. yulia.worbe@aphp.fr.
Department of Neurology, Clinical Investigation Center for Neurosciences, Assistance Publique-Hôpitaux de Paris, Pitié-Salpêtrière Hospital, Paris, France. yulia.worbe@aphp.fr.
Department of Neurophysiology, Saint-Antoine Hospital, Paris, France. yulia.worbe@aphp.fr.

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