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Clinical relevance of the TECTA c.6183G>T variant identified in a family with autosomal dominant hearing loss: a case report
Missense variants in the α-tectorin gene (TECTA) cause autosomal dominant (DFNA8/A12) non-syndromic hearing loss (ADNSHL) and account for a considerable number of ADNSHL cases. According to genotype-phenotype correlation studies, missense variants in the zona pellucida (ZP) domain of α-tectorin pred...
Autores principales: | , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Croatian Medical Schools
2023
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10668043/ https://www.ncbi.nlm.nih.gov/pubmed/37927186 http://dx.doi.org/10.3325/cmj.2023.64.329 |
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author | Sansović, Ivona Meašić, Ana-Maria Odak, Ljubica Kero, Mijana |
author_facet | Sansović, Ivona Meašić, Ana-Maria Odak, Ljubica Kero, Mijana |
author_sort | Sansović, Ivona |
collection | PubMed |
description | Missense variants in the α-tectorin gene (TECTA) cause autosomal dominant (DFNA8/A12) non-syndromic hearing loss (ADNSHL) and account for a considerable number of ADNSHL cases. According to genotype-phenotype correlation studies, missense variants in the zona pellucida (ZP) domain of α-tectorin predominantly cause mid-frequency HL. Here, we report on clinical exome sequencing results in a large family with early-onset, sensorineural, moderate-to-severe mid-frequency HL. We identified one heterozygous c.6183G>T variant near the ZP domain of TECTA segregating in five family members. This variant was previously reported as a variant of uncertain significance in a family with ADNSHL. On the basis of specific segregation in the currently studied family and the general guidelines of the American College of Medical Genetics and Genomics, we argue that the TECTA c.6183G>T variant should be considered a likely pathogenic cause of ADNSHL. This report adds to the knowledge on the rare c.6183G>T missense variant, which affects the immediate vicinity of the ZP domain in TECTA. Our findings highlight the importance of clinical evaluation in patients with familial HL and of studying family segregation when assessing the pathogenicity of a variant. |
format | Online Article Text |
id | pubmed-10668043 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | Croatian Medical Schools |
record_format | MEDLINE/PubMed |
spelling | pubmed-106680432023-10-01 Clinical relevance of the TECTA c.6183G>T variant identified in a family with autosomal dominant hearing loss: a case report Sansović, Ivona Meašić, Ana-Maria Odak, Ljubica Kero, Mijana Croat Med J Case Report Missense variants in the α-tectorin gene (TECTA) cause autosomal dominant (DFNA8/A12) non-syndromic hearing loss (ADNSHL) and account for a considerable number of ADNSHL cases. According to genotype-phenotype correlation studies, missense variants in the zona pellucida (ZP) domain of α-tectorin predominantly cause mid-frequency HL. Here, we report on clinical exome sequencing results in a large family with early-onset, sensorineural, moderate-to-severe mid-frequency HL. We identified one heterozygous c.6183G>T variant near the ZP domain of TECTA segregating in five family members. This variant was previously reported as a variant of uncertain significance in a family with ADNSHL. On the basis of specific segregation in the currently studied family and the general guidelines of the American College of Medical Genetics and Genomics, we argue that the TECTA c.6183G>T variant should be considered a likely pathogenic cause of ADNSHL. This report adds to the knowledge on the rare c.6183G>T missense variant, which affects the immediate vicinity of the ZP domain in TECTA. Our findings highlight the importance of clinical evaluation in patients with familial HL and of studying family segregation when assessing the pathogenicity of a variant. Croatian Medical Schools 2023-10 /pmc/articles/PMC10668043/ /pubmed/37927186 http://dx.doi.org/10.3325/cmj.2023.64.329 Text en Copyright © 2023 by the Croatian Medical Journal. All rights reserved. https://creativecommons.org/licenses/by/2.5/This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. |
spellingShingle | Case Report Sansović, Ivona Meašić, Ana-Maria Odak, Ljubica Kero, Mijana Clinical relevance of the TECTA c.6183G>T variant identified in a family with autosomal dominant hearing loss: a case report |
title | Clinical relevance of the TECTA c.6183G>T variant identified in a family with autosomal dominant hearing loss: a case report |
title_full | Clinical relevance of the TECTA c.6183G>T variant identified in a family with autosomal dominant hearing loss: a case report |
title_fullStr | Clinical relevance of the TECTA c.6183G>T variant identified in a family with autosomal dominant hearing loss: a case report |
title_full_unstemmed | Clinical relevance of the TECTA c.6183G>T variant identified in a family with autosomal dominant hearing loss: a case report |
title_short | Clinical relevance of the TECTA c.6183G>T variant identified in a family with autosomal dominant hearing loss: a case report |
title_sort | clinical relevance of the tecta c.6183g>t variant identified in a family with autosomal dominant hearing loss: a case report |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10668043/ https://www.ncbi.nlm.nih.gov/pubmed/37927186 http://dx.doi.org/10.3325/cmj.2023.64.329 |
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