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Epidermolysis Bullosa in Chinese Patients: Genetic Analysis and Mutation Landscape in 57 Pedigrees and Sporadic Cases
Epidermolysis bullosa encompasses a group of inherited blistering skin disorders. The pathogenic mutations in 10–25% of patients with epidermolysis bullosa have not been identified by Sanger sequencing. The aims of this study were to identify the pathogenic sequence alterations in a large cohort of...
Autores principales: | , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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Society for Publication of Acta Dermato-Venereologica
2021
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9413781/ https://www.ncbi.nlm.nih.gov/pubmed/34046686 http://dx.doi.org/10.2340/00015555-3843 |
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author | YU, Yueqian WANG, Zhenzhen MI, Zihao SUN, Lele FU, Xi’an YU, Gongqi PANG, Zheng LIU, Hong ZHANG, Furen |
author_facet | YU, Yueqian WANG, Zhenzhen MI, Zihao SUN, Lele FU, Xi’an YU, Gongqi PANG, Zheng LIU, Hong ZHANG, Furen |
author_sort | YU, Yueqian |
collection | PubMed |
description | Epidermolysis bullosa encompasses a group of inherited blistering skin disorders. The pathogenic mutations in 10–25% of patients with epidermolysis bullosa have not been identified by Sanger sequencing. The aims of this study were to identify the pathogenic sequence alterations in a large cohort of Chinese patients with epidermolysis bullosa and to clarify the relationship between clinical phenotypes and genotypes. Whole-exome sequencing was performed on 44 pedigrees and 13 sporadic cases. The results were further confirmed by Sanger sequencing. In total, 52 mutations, comprising 19 novel and 33 previously reported mutations, were identified in 5 genes, with a mutation detection rate of 100%. A relationship between subtypes and pathogenic genes was established: 12 cases of epidermolysis bullosa simplex were associated with mutations in KRT5/14 and PLEC; one case of junctional epidermolysis bullosa carried mutations in ITGB4; and 44 cases of dystrophic epidermolysis bullosa were caused by mutations in COL7A1. The results of this study support whole-exome sequencing as a promising tool in the genetic diagnosis of epidermolysis bullosa. |
format | Online Article Text |
id | pubmed-9413781 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2021 |
publisher | Society for Publication of Acta Dermato-Venereologica |
record_format | MEDLINE/PubMed |
spelling | pubmed-94137812022-10-20 Epidermolysis Bullosa in Chinese Patients: Genetic Analysis and Mutation Landscape in 57 Pedigrees and Sporadic Cases YU, Yueqian WANG, Zhenzhen MI, Zihao SUN, Lele FU, Xi’an YU, Gongqi PANG, Zheng LIU, Hong ZHANG, Furen Acta Derm Venereol Investigative Report Epidermolysis bullosa encompasses a group of inherited blistering skin disorders. The pathogenic mutations in 10–25% of patients with epidermolysis bullosa have not been identified by Sanger sequencing. The aims of this study were to identify the pathogenic sequence alterations in a large cohort of Chinese patients with epidermolysis bullosa and to clarify the relationship between clinical phenotypes and genotypes. Whole-exome sequencing was performed on 44 pedigrees and 13 sporadic cases. The results were further confirmed by Sanger sequencing. In total, 52 mutations, comprising 19 novel and 33 previously reported mutations, were identified in 5 genes, with a mutation detection rate of 100%. A relationship between subtypes and pathogenic genes was established: 12 cases of epidermolysis bullosa simplex were associated with mutations in KRT5/14 and PLEC; one case of junctional epidermolysis bullosa carried mutations in ITGB4; and 44 cases of dystrophic epidermolysis bullosa were caused by mutations in COL7A1. The results of this study support whole-exome sequencing as a promising tool in the genetic diagnosis of epidermolysis bullosa. Society for Publication of Acta Dermato-Venereologica 2021-07-15 /pmc/articles/PMC9413781/ /pubmed/34046686 http://dx.doi.org/10.2340/00015555-3843 Text en © 2021 Acta Dermato-Venereologica https://creativecommons.org/licenses/by-nc/4.0/This is an open access article under the CC BY-NC license |
spellingShingle | Investigative Report YU, Yueqian WANG, Zhenzhen MI, Zihao SUN, Lele FU, Xi’an YU, Gongqi PANG, Zheng LIU, Hong ZHANG, Furen Epidermolysis Bullosa in Chinese Patients: Genetic Analysis and Mutation Landscape in 57 Pedigrees and Sporadic Cases |
title | Epidermolysis Bullosa in Chinese Patients: Genetic Analysis and Mutation Landscape in 57 Pedigrees and Sporadic Cases |
title_full | Epidermolysis Bullosa in Chinese Patients: Genetic Analysis and Mutation Landscape in 57 Pedigrees and Sporadic Cases |
title_fullStr | Epidermolysis Bullosa in Chinese Patients: Genetic Analysis and Mutation Landscape in 57 Pedigrees and Sporadic Cases |
title_full_unstemmed | Epidermolysis Bullosa in Chinese Patients: Genetic Analysis and Mutation Landscape in 57 Pedigrees and Sporadic Cases |
title_short | Epidermolysis Bullosa in Chinese Patients: Genetic Analysis and Mutation Landscape in 57 Pedigrees and Sporadic Cases |
title_sort | epidermolysis bullosa in chinese patients: genetic analysis and mutation landscape in 57 pedigrees and sporadic cases |
topic | Investigative Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9413781/ https://www.ncbi.nlm.nih.gov/pubmed/34046686 http://dx.doi.org/10.2340/00015555-3843 |
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