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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...

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Autores principales: YU, Yueqian, WANG, Zhenzhen, MI, Zihao, SUN, Lele, FU, Xi’an, YU, Gongqi, PANG, Zheng, LIU, Hong, ZHANG, Furen
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Society for Publication of Acta Dermato-Venereologica 2021
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.
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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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