Evidence map›Paper›PMID 40100472›Full record

ArticleEuropean journal of pediatrics2025

Whole exome sequencing diagnosing syndromic and non-syndromic hearing loss with expansion of the phenotypic spectrum related to TMC1 variants.

Nagham M Elbagoury, Engy A Ashaat, Mona K Mekkawy, Ragaey Y Mohamed, Anas M Askoura, Peter M Milad, Mona L Essawi

Abstract read
In one paragraph

Article in European journal of pediatrics, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

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3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

7 authors.

Nagham M ElbagouryDepartment of Medical Molecular Genetics, National Research Centre, Giza, 12311, Egypt. naghamelbagoury@hotmail.com.
Engy A AshaatDepartment of Clinical Genetics, National R esearch Centre, Giza, Egypt.
Mona K MekkawyDepartment of Human Cytogenetics, National Research Center, Giza, Egypt.
Ragaey Y MohamedDepartment of Otorhinolaryngology, Faculty of Medicine, Beni-Suef University, Beni-Suef, Egypt.
Anas M AskouraDepartment of Otorhinolaryngology, Faculty of Medicine, Ain Shams University, Cairo, Egypt.
Peter M MiladDepartment of Otorhinolaryngology, Faculty of Medicine, Ain Shams University, Cairo, Egypt.
Mona L EssawiDepartment of Medical Molecular Genetics, National Research Centre, Giza, 12311, Egypt.

Funding

National Research Centre 12060187
6 · The paper itself

Abstract

Hearing loss (HL) is an impending disorder. The high incidence of congenital genetic HL affects the language and communication skills of a large number of children worldwide. Our study is mainly concerned with exploring the genetic etiology of congenital hearing loss through Sanger sequencing of the coding exon in GJB2, the most common causative gene worldwide, in 17 patients from 13 unrelated families followed by whole exome sequencing for cases showing biallelic wildtype GJB2. Eleven patients from eight families showed homozygous and compound heterozygous variants in the GJB2 gene. Six patients from five families proceeded to whole exome sequencing. One of them showed a reported variant in ILDR1, and three showed novel variants in the TMC1 and KCNQ1 genes. Two showed variants reported for the first time in HL patients in the PEX6 and MYO3A genes.In conclusion, this study suggests new insights into the contribution of MYO3A, KCNQ1, and PEX6 to congenital sensorineural hearing loss as well as possible expansion of the phenotypic spectrum of the TMC1 gene. What is Known: • Sanger sequencing and whole exome sequencing are used for molecular diagnosis of syndromic and non-syndromic types of hearing loss (HL). • TMC1 gene causes a type of non-syndromic HL. What is New: • Expanding the molecular spectrum of MYO3A, PEX6, TMC1, and KCNQ1 genes as contributor genes in HL by detecting variants first time to be detected in HL patients. • Expanding the clinical spectrum of TMC1 gene to cause syndromic and non-syndromic HL.

Indexed as

Exome SequencingHearing LossHearing Loss, SensorineuralMembrane ProteinsAdolescentChildChild, PreschoolConnexin 26ConnexinsFemaleHumansInfantMaleMutationPedigreePhenotypeConnexin 26ConnexinsGJB2 protein, humanMembrane ProteinsTMC1 protein, humanGJB2ILDR1KCNQ1MYO3APEX6SNHL

Identifiers

PMID40100472
PMCPMC11919965

What Socratic holds

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LicenceCC BY
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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the Socratic graph.