Muscular phenotype description of abnormal THOC2 splicing.

Cytoplasmic body Fetal arthrogryposis Myopathy THOC2 mutation

Journal

Neuromuscular disorders : NMD
ISSN: 1873-2364
Titre abrégé: Neuromuscul Disord
Pays: England
ID NLM: 9111470

Informations de publication

Date de publication:
Dec 2023
Historique:
received: 02 08 2023
revised: 20 09 2023
accepted: 26 09 2023
pubmed: 10 11 2023
medline: 10 11 2023
entrez: 9 11 2023
Statut: ppublish

Résumé

Until recently, the disease known to be associated with THOC2 mutations was Intellectual developmental disorder, X-linked 12 (MIM300957). However, recently, fetal arthrogryposis multiplex congenita has been associated with a specific splice site mutation in the THOC2 gene. We report a family with the same splice site mutation in the THOC2 gene involved in fetal arthrogryposis as well. We provide the first description of the muscular phenotype of this disease which reveals the presence of cytoplasmic bodies. Our findings expand the clinical phenotype of THOC2 gene related defects.

Identifiants

pubmed: 37945483
pii: S0960-8966(23)00735-6
doi: 10.1016/j.nmd.2023.09.009
pii:
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

978-982

Informations de copyright

Copyright © 2023. Published by Elsevier B.V.

Déclaration de conflit d'intérêts

Declaration of Competing Interest The authors declare that they have no competing interests.

Auteurs

Charlotte Dubucs (C)

Pathology Department, Institut Universitaire du cancer de Toulouse, Toulouse, France; Genetic Medical Department, Toulouse University Hospital, Toulouse, France. Electronic address: Dubucs.charlotte@iuct-oncopole.fr.

John Rendu (J)

Grenoble Alpes University, Inserm, U1216, Grenoble Alpes University Hospital, Grenoble Institut Neurosciences, 38000 Grenoble, France.

Laurence Michel-Calemard (L)

Service Biochimie et Biologie Moléculaire - Pathologies endocriniennes rénales, musculaires et mucoviscidose, Centre de Biologie et Pathologie Est, CHU de Lyon HCL - GH Est, France.

Rita Menassa (R)

Service Biochimie et Biologie Moléculaire - Pathologies endocriniennes rénales, musculaires et mucoviscidose, Centre de Biologie et Pathologie Est, CHU de Lyon HCL - GH Est, France.

Maud Langeois (M)

Genetic Medical Department, Toulouse University Hospital, Toulouse, France.

Yvan Nicaise (Y)

INSERM U1037, Cancer Research Center of Toulouse (CRCT), Toulouse, France.

Jessie Ousselin (J)

Pathology Department, Institut Universitaire du cancer de Toulouse, Toulouse, France.

Jacqueline Aziza (J)

Pathology Department, Institut Universitaire du cancer de Toulouse, Toulouse, France.

Emmanuelle Uro-Coste (E)

Pathology Department, Institut Universitaire du cancer de Toulouse, Toulouse, France.

Classifications MeSH