title: Orthostatic tremor: a cerebellar pathology?
abstract: SEE MUTHURAMAN ET AL DOI101093/AWW164 FOR A SCIENTIFIC COMMENTARY ON THIS ARTICLE: Primary orthostatic tremor is characterized by high frequency tremor affecting the legs and trunk during the standing position. Cerebellar defects were suggested in orthostatic tremor without direct evidence. We aimed to characterize the anatomo-functional defects of the cerebellar motor pathways in orthostatic tremor. We used multimodal neuroimaging to compare 17 patients with orthostatic tremor and 17 age- and gender-matched healthy volunteers. Nine of the patients with orthostatic tremor underwent repetitive transcranial stimulation applied over the cerebellum during five consecutive days. We quantified the duration of standing position and tremor severity through electromyographic recordings. Compared to healthy volunteers, grey matter volume in patients with orthostatic tremor was (i) increased in the cerebellar vermis and correlated positively with the duration of the standing position; and (ii) increased in the supplementary motor area and decreased in the lateral cerebellum, which both correlated with the disease duration. Functional connectivity between the lateral cerebellum and the supplementary motor area was abnormally increased in patients with orthostatic tremor, and correlated positively with tremor severity. After repetitive transcranial stimulation, tremor severity and functional connectivity between the lateral cerebellum and the supplementary motor area were reduced. We provide an explanation for orthostatic tremor pathophysiology, and demonstrate the functional relevance of cerebello-thalamo-cortical connections in tremor related to cerebellar defects.
authors.affiliation: 1 Centre de NeuroImagerie de Recherche - Institut du Cerveau et de la Moelle épinière, ICM, Paris, France 2 Sorbonne Universités, UPMC Univ Paris 06, UMR S 1127, Paris, France 3 CNRS, UMR 7225, Paris, France 4 Inserm, U 1127, Paris, France 5 AP-HP, Hôpital de la Pitié Salpêtrière, Département de Neuroradiologie, Paris, France felix.cervantes@ars.usda.gov.
authors.forename: Cécile
authors.initials: C
authors.lastname: Gallea
authors.affiliation: 1 Centre de NeuroImagerie de Recherche - Institut du Cerveau et de la Moelle épinière, ICM, Paris, France 2 Sorbonne Universités, UPMC Univ Paris 06, UMR S 1127, Paris, France 3 CNRS, UMR 7225, Paris, France 4 Inserm, U 1127, Paris, France.
authors.forename: Traian
authors.initials: T
authors.lastname: Popa
authors.affiliation: 1 Centre de NeuroImagerie de Recherche - Institut du Cerveau et de la Moelle épinière, ICM, Paris, France 2 Sorbonne Universités, UPMC Univ Paris 06, UMR S 1127, Paris, France 3 CNRS, UMR 7225, Paris, France 4 Inserm, U 1127, Paris, France 5 AP-HP, Hôpital de la Pitié Salpêtrière, Département de Neuroradiologie, Paris, France.
authors.forename: Daniel
authors.initials: D
authors.lastname: García-Lorenzo
authors.affiliation: 1 Centre de NeuroImagerie de Recherche - Institut du Cerveau et de la Moelle épinière, ICM, Paris, France 2 Sorbonne Universités, UPMC Univ Paris 06, UMR S 1127, Paris, France 3 CNRS, UMR 7225, Paris, France 4 Inserm, U 1127, Paris, France 5 AP-HP, Hôpital de la Pitié Salpêtrière, Département de Neuroradiologie, Paris, France.
authors.forename: Romain
authors.initials: R
authors.lastname: Valabregue
authors.affiliation: 6 ESPCI, Paris Sciences, Paris, France.
authors.forename: André-Pierre
authors.initials: AP
authors.lastname: Legrand
authors.affiliation: 2 Sorbonne Universités, UPMC Univ Paris 06, UMR S 1127, Paris, France 3 CNRS, UMR 7225, Paris, France 4 Inserm, U 1127, Paris, France 7 AP-HP, Hôpital de Saint-Antoine, Département de Neurologie, Paris, France.
authors.forename: Emmanuelle
authors.initials: E
authors.lastname: Apartis
authors.affiliation: 1 Centre de NeuroImagerie de Recherche - Institut du Cerveau et de la Moelle épinière, ICM, Paris, France 2 Sorbonne Universités, UPMC Univ Paris 06, UMR S 1127, Paris, France 3 CNRS, UMR 7225, Paris, France 4 Inserm, U 1127, Paris, France 5 AP-HP, Hôpital de la Pitié Salpêtrière, Département de Neuroradiologie, Paris, France.
authors.forename: Lea
authors.initials: L
authors.lastname: Marais
authors.affiliation: 2 Sorbonne Universités, UPMC Univ Paris 06, UMR S 1127, Paris, France 3 CNRS, UMR 7225, Paris, France 4 Inserm, U 1127, Paris, France 8 AP-HP, Hôpital de la Pitié Salpêtrière, Département de Neurologie, Paris, France.
authors.forename: Bertrand
authors.initials: B
authors.lastname: Degos
authors.affiliation: 2 Sorbonne Universités, UPMC Univ Paris 06, UMR S 1127, Paris, France 3 CNRS, UMR 7225, Paris, France 4 Inserm, U 1127, Paris, France 8 AP-HP, Hôpital de la Pitié Salpêtrière, Département de Neurologie, Paris, France.
authors.forename: Cecile
authors.initials: C
authors.lastname: Hubsch
authors.affiliation: 1 Centre de NeuroImagerie de Recherche - Institut du Cerveau et de la Moelle épinière, ICM, Paris, France 2 Sorbonne Universités, UPMC Univ Paris 06, UMR S 1127, Paris, France 3 CNRS, UMR 7225, Paris, France 4 Inserm, U 1127, Paris, France.
authors.forename: Sara
authors.initials: S
authors.lastname: Fernández-Vidal
authors.affiliation: 1 Centre de NeuroImagerie de Recherche - Institut du Cerveau et de la Moelle épinière, ICM, Paris, France 2 Sorbonne Universités, UPMC Univ Paris 06, UMR S 1127, Paris, France 3 CNRS, UMR 7225, Paris, France 4 Inserm, U 1127, Paris, France.
authors.forename: Eric
authors.initials: E
authors.lastname: Bardinet
authors.affiliation: 2 Sorbonne Universités, UPMC Univ Paris 06, UMR S 1127, Paris, France 3 CNRS, UMR 7225, Paris, France 4 Inserm, U 1127, Paris, France 8 AP-HP, Hôpital de la Pitié Salpêtrière, Département de Neurologie, Paris, France.
authors.forename: Emmanuel
authors.initials: E
authors.lastname: Roze
authors.affiliation: 1 Centre de NeuroImagerie de Recherche - Institut du Cerveau et de la Moelle épinière, ICM, Paris, France 2 Sorbonne Universités, UPMC Univ Paris 06, UMR S 1127, Paris, France 3 CNRS, UMR 7225, Paris, France 4 Inserm, U 1127, Paris, France 5 AP-HP, Hôpital de la Pitié Salpêtrière, Département de Neuroradiologie, Paris, France.
authors.forename: Stéphane
authors.initials: S
authors.lastname: Lehéricy
authors.affiliation: 2 Sorbonne Universités, UPMC Univ Paris 06, UMR S 1127, Paris, France 3 CNRS, UMR 7225, Paris, France 4 Inserm, U 1127, Paris, France.
authors.forename: Sabine
authors.initials: S
authors.lastname: Meunier
authors.affiliation: 2 Sorbonne Universités, UPMC Univ Paris 06, UMR S 1127, Paris, France 3 CNRS, UMR 7225, Paris, France 4 Inserm, U 1127, Paris, France 8 AP-HP, Hôpital de la Pitié Salpêtrière, Département de Neurologie, Paris, France.
authors.forename: Marie
authors.initials: M
authors.lastname: Vidailhet
mesh_terms: D000328:Adult; D000368:Aged; D002526:Cerebellar Diseases / Q000000981:diagnostic imaging / Q000503:physiopathology / Q000628:therapy; D004244:Dizziness / Q000000981:diagnostic imaging / Q000503:physiopathology / Q000628:therapy; D004525:Efferent Pathways; D004576:Electromyography; D005260:Female; D059907:Functional Neuroimaging / Q000379:methods*; D006801:Humans; D008279:Magnetic Resonance Imaging; D008297:Male; D008875:Middle Aged; D009044:Motor Cortex / Q000000981:diagnostic imaging*; D009415:Nerve Net / Q000000981:diagnostic imaging / Q000503:physiopathology*; D050781:Transcranial Magnetic Stimulation / Q000379:methods*; D016896:Treatment Outcome; D014202:Tremor / Q000000981:diagnostic imaging / Q000503:physiopathology / Q000628:therapy
keywords: cerebellar function; frontal lobe; motor cortex; movement disorder; tremor