The p.Gly130Val mutation in the GJB2 gene: A familiar case of autosomal dominant non-syndromic hearing loss.


Journal

International journal of pediatric otorhinolaryngology
ISSN: 1872-8464
Titre abrégé: Int J Pediatr Otorhinolaryngol
Pays: Ireland
ID NLM: 8003603

Informations de publication

Date de publication:
Dec 2019
Historique:
received: 09 03 2019
revised: 01 08 2019
accepted: 20 08 2019
pubmed: 1 9 2019
medline: 8 2 2020
entrez: 1 9 2019
Statut: ppublish

Résumé

Several forms of sensorineural hearing loss (SNHL) have been imputated to connexins mutations and prevalently to connexin 26 (Cx26), codified by the GJB2 gene (gap junction protein, beta 2). Here, we report the first familiar case (heterozygous p. G130V mutation) of non-syndromic (without any dermatological manifestation) dominant profound SNHL. Proband was a 6-years-old male with post-lingual bilateral profound SNHL, clinically identified at the age of 3 with diagnosis of severe SNHL. We confirm that the p. G130V variant of the GJB2 gene is causative of autosomal dominant form of SNHL, although it is not always associated with the presence of skin diseases.

Identifiants

pubmed: 31472357
pii: S0165-5876(19)30397-0
doi: 10.1016/j.ijporl.2019.109653
pii:
doi:

Substances chimiques

Connexins 0
GJB2 protein, human 0
Connexin 26 127120-53-0

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

109653

Informations de copyright

Copyright © 2019 Elsevier B.V. All rights reserved.

Auteurs

Adelaide Bussini (A)

Unit of Cytogenetics and Medical Genetic, ASST Settelaghi, Varese, Italy.

Rossana Righi (R)

Unit of Cytogenetics and Medical Genetic, ASST Settelaghi, Varese, Italy.

Chiara Pessina (C)

Unit of Cytogenetics and Medical Genetic, ASST Settelaghi, Varese, Italy.

Angelo Genoni (A)

Unit of Cytogenetics and Medical Genetic, ASST Settelaghi, Varese, Italy.

Eliana Cristofari (E)

Unit of Audiology, ASST Settelaghi, Varese, Italy.

Annalisa Meli (A)

Unit of Audiology, ASST Settelaghi, Varese, Italy.

Paola Granata (P)

Unit of Cytogenetics and Medical Genetic, ASST Settelaghi, Varese, Italy.

Emanuela Meroni (E)

Unit of Cytogenetics and Medical Genetic, ASST Settelaghi, Varese, Italy.

Francesco Broccolo (F)

Department of Medicine and Surgery (School of Medicine), University of Milano-Bicocca, Monza, Italy. Electronic address: francesco.broccolo@unimib.it.

Rosario Casalone (R)

Unit of Cytogenetics and Medical Genetic, ASST Settelaghi, Varese, Italy. Electronic address: rosario.casalone@asst-settelaghi.it.

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Classifications MeSH