LHFPL5 mutation: A rare cause of non-syndromic autosomal recessive hearing loss.


Journal

European journal of medical genetics
ISSN: 1878-0849
Titre abrégé: Eur J Med Genet
Pays: Netherlands
ID NLM: 101247089

Informations de publication

Date de publication:
Dec 2019
Historique:
received: 08 02 2018
revised: 30 07 2018
accepted: 22 11 2018
pubmed: 27 11 2018
medline: 13 3 2020
entrez: 27 11 2018
Statut: ppublish

Résumé

Hearing loss is a debilitating disorder that impairs language acquisition, resulting in disability in children and potential isolation in adulthood. Its onset can have a genetic basis, though environmental factors, which are often preventable, can also cause the condition. The genetic forms are highly heterogeneous, and early detection is necessary to arrange appropriate patient support. Here we report the molecular basis of hereditary hearing loss in a consanguineous family with multiple affected members from Oman. Combining homozygosity mapping with whole exome sequencing identified a novel homozygous nucleotide substitution c.575T > C in the lipoma HMGIC fusion partner-like 5 gene (LHFPL5), that converted the 192

Identifiants

pubmed: 30476627
pii: S1769-7212(18)30102-2
doi: 10.1016/j.ejmg.2018.11.026
pii:
doi:

Substances chimiques

LHFPL5 protein, human 0
Membrane Proteins 0

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

103592

Subventions

Organisme : Medical Research Council
ID : MR/J004391/1
Pays : United Kingdom

Informations de copyright

Crown Copyright © 2018. Published by Elsevier Masson SAS. All rights reserved.

Auteurs

Ahmed H Al-Amri (AH)

Section of Ophthalmology & Neuroscience, Leeds Institute of Medical Research at St. James's, University of Leeds, United Kingdom; School of Biomedical Sciences, University of Leeds, United Kingdom; National Genetic Centre, Directorate General of Royal Hospital, Ministry of Health, Muscat, Oman.

Abeer Al Saegh (A)

Genetic & Developmental Medicine Clinic, Genetics Department, Sultan Qaboos University Hospital, Muscat, Oman.

Watfa Al-Mamari (W)

Developmental Pediatric Unit, Child Health Department, Sultan Qaboos University Hospital, Muscat, Oman.

Mohammed E El-Asrag (ME)

Section of Ophthalmology & Neuroscience, Leeds Institute of Medical Research at St. James's, University of Leeds, United Kingdom; Department of Zoology, Faculty of Science, Benha University, Benha, Egypt.

Mohammed N Al-Kindi (MN)

Biochemistry Department, Sultan Qaboos University Hospital, Muscat, Oman.

Mazin Al Khabouri (M)

Department of Ear, Nose & Throat, Al Nahdha Hospital, Ministry of Health, Muscat, Oman.

Nadia Al Wardy (N)

Biochemistry Department, Sultan Qaboos University Hospital, Muscat, Oman.

Khalsa Al Lamki (K)

Biochemistry Department, Sultan Qaboos University Hospital, Muscat, Oman.

Ahlam Gabr (A)

Developmental Pediatric Unit, Child Health Department, Sultan Qaboos University Hospital, Muscat, Oman.

Ahmed Idris (A)

Developmental Pediatric Unit, Child Health Department, Sultan Qaboos University Hospital, Muscat, Oman.

Chris F Inglehearn (CF)

Section of Ophthalmology & Neuroscience, Leeds Institute of Medical Research at St. James's, University of Leeds, United Kingdom.

Steven J Clapcote (SJ)

School of Biomedical Sciences, University of Leeds, United Kingdom.

Manir Ali (M)

Section of Ophthalmology & Neuroscience, Leeds Institute of Medical Research at St. James's, University of Leeds, United Kingdom. Electronic address: m.ali@leeds.ac.uk.

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Classifications MeSH