ELMOD3-SH2D6 gene fusion as a possible co-star actor in autism spectrum disorder scenario.


Journal

Journal of cellular and molecular medicine
ISSN: 1582-4934
Titre abrégé: J Cell Mol Med
Pays: England
ID NLM: 101083777

Informations de publication

Date de publication:
01 2020
Historique:
received: 30 07 2019
revised: 04 09 2019
accepted: 07 09 2019
pubmed: 5 12 2019
medline: 23 4 2021
entrez: 5 12 2019
Statut: ppublish

Résumé

Autism spectrum disorder (ASD) is a group of neurodevelopmental disorders characterized by high heritability. It is known that genetic factors contribute to ASD pathogenesis. In particular, copy number variants (CNVs) are involved in ASD susceptibility and can affect gene expression regulation. 2p11.2 microdeletions encompassing ELMOD3, CAPG and SH2D6 genes have been described in four unrelated ASD families. The present study revealed that this microdeletion is responsible for the production of a chimeric transcript generated from the fusion between ELMOD3 and SH2D6. The identified transcript showed significantly higher expression levels in subjects carrying the deletion compared to control subjects, suggesting that it is not subjected to nonsense-mediated decay and might encode for a chimeric protein. In conclusion, this study suggests the possible involvement of this gene fusion, together with the other previously identified variants, in ASD.

Identifiants

pubmed: 31800155
doi: 10.1111/jcmm.14733
pmc: PMC6991669
doi:

Substances chimiques

ELMOD3 protein, human 0
GTPase-Activating Proteins 0
Intracellular Signaling Peptides and Proteins 0
SH2D6 protein, human 0

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

2064-2069

Informations de copyright

© 2019 The Authors. Journal of Cellular and Molecular Medicine published by John Wiley & Sons Ltd and Foundation for Cellular and Molecular Medicine.

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Auteurs

Eleonora Loi (E)

Department of Biomedical Sciences, Unit of Biology and Genetics, University of Cagliari, Cagliari, Italy.

Loredana Moi (L)

Department of Biomedical Sciences, Unit of Biology and Genetics, University of Cagliari, Cagliari, Italy.

Sylvain Blois (S)

Department of Biomedical Sciences, Unit of Biology and Genetics, University of Cagliari, Cagliari, Italy.

Elena Bacchelli (E)

Department of Pharmacy and Biotechnology, University of Bologna, Bologna, Italy.

Ana Florencia Vega Benedetti (AF)

Department of Biomedical Sciences, Unit of Biology and Genetics, University of Cagliari, Cagliari, Italy.

Cinzia Cameli (C)

Department of Pharmacy and Biotechnology, University of Bologna, Bologna, Italy.

Roberta Fadda (R)

Department of Pedagogy, Psychology, Philosophy, University of Cagliari, Cagliari, Italy.

Elena Maestrini (E)

Department of Pharmacy and Biotechnology, University of Bologna, Bologna, Italy.

Marinella Carta (M)

Center for Pervasive Developmental Disorders, AO Brotzu, Cagliari, Italy.

Giuseppe Doneddu (G)

Center for Pervasive Developmental Disorders, AO Brotzu, Cagliari, Italy.

Patrizia Zavattari (P)

Department of Biomedical Sciences, Unit of Biology and Genetics, University of Cagliari, Cagliari, Italy.

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Classifications MeSH