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Loss of paraplegin drives spasticity rather than ataxia in a cohort of 241 patients with SPG7

Authors :
Ludger Schöls
Isabelle Migeotte
Claire Sophie Davoine
Mélanie Papin
Pegah Masrori
Bart P.C. van de Warrenburg
Rebecca Schüle
Mathieu Anheim
Marc D'Hooghe
Elisabeth Ollagnon-Roman
Charles Duyckaerts
Claire Ewenczyk
Andrea Martinuzzi
Jonathan Baets
Marie Lorraine Monin
Giulia Coarelli
T Deconinck
Maria Grazia D'Angelo
Sophie Tezenas du Montcel
Alexis Brice
Thomas Klockgether
Delia Kurzwelly
Fanny Mochel
Perrine Charles
Peter De Jonghe
Elisa E.G. Hamer
Matthis Synofzik
B. Fontaine
Maria Teresa Bassi
Christoph Kamm
Danielle Seilhean
Giovanni Stevanin
Thomas Klopstock
Jan De Bleecker
Alexandra Durr
Guillaume Banneau
Institut du Cerveau et de la Moëlle Epinière = Brain and Spine Institute (ICM)
Institut National de la Santé et de la Recherche Médicale (INSERM)-CHU Pitié-Salpêtrière [AP-HP]
Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU)-Sorbonne Université (SU)-Centre National de la Recherche Scientifique (CNRS)
Service de Neurochirurgie [CHU Pitié-Salpêtrière]
CHU Pitié-Salpêtrière [AP-HP]
Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU)
Hertie-Institute for Clinical Brain Research
Département de Neurologie
CHU Strasbourg
Centre de recherche en Myologie – U974 SU-INSERM
Institut National de la Santé et de la Recherche Médicale (INSERM)-Sorbonne Université (SU)
Munich Cluster for systems neurology [Munich] (SyNergy)
Technische Universität Munchen - Université Technique de Munich [Munich, Allemagne] (TUM)-Ludwig-Maximilians-Universität München (LMU)
Hôpital de la Croix-Rousse [CHU - HCL]
Hospices Civils de Lyon (HCL)
École pratique des hautes études (EPHE)
Université Paris sciences et lettres (PSL)
Département de Biostatistique, Santé Publique et Information Médicale [CHU Pitié-Salpêtrière] (BIOSPIM )
Institut Pierre Louis d'Epidémiologie et de Santé Publique (iPLESP)
Sorbonne Université (SU)-Institut National de la Santé et de la Recherche Médicale (INSERM)
Service de Neuropathologie [CHU Pitié Salpêtrière]
Laboratoire de Neuropathologie Raymond Escourolle
Centre de Recherche en Myologie
Source :
Neurology, Neurology, 92, e2679-e2690, Neurology, American Academy of Neurology, 2019, 92 (23), pp.e2679-e2690. ⟨10.1212/WNL.0000000000007606⟩, Neurology 92(23), e2679-e2690 (2019). doi:10.1212/WNL.0000000000007606, Neurology, 92, 23, pp. e2679-e2690, Neurology, 92 (23
Publication Year :
2019

Abstract

Objective: We took advantage of a large multinational recruitment to delineate genotype-phenotype correlations in a large, trans-European multicenter cohort of patients with spastic paraplegia gene 7 (SPG7). Methods: We analyzed clinical and genetic data from 241 patients with SPG7, integrating neurologic follow-up data. One case was examined neuropathologically. Results: Patients with SPG7 had a mean age of 35.5 ± 14.3 years (n = 233) at onset and presented with spasticity (n = 89), ataxia (n = 74), or both (n = 45). At the first visit, patients with a longer disease duration (>20 years, n = 62) showed more cerebellar dysarthria (p < 0.05), deep sensory loss (p < 0.01), muscle wasting (p < 0.01), ophthalmoplegia (p < 0.05), and sphincter dysfunction (p < 0.05) than those with a shorter duration (<br />SCOPUS: ar.j<br />info:eu-repo/semantics/published

Details

Language :
English
ISSN :
00283878 and 1526632X
Database :
OpenAIRE
Journal :
Neurology
Accession number :
edsair.doi.dedup.....ad13a0fe5a53812528a1024662571ae0
Full Text :
https://doi.org/10.1212/WNL.0000000000007606⟩