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A Novel Mutation in the FYCO1 Gene Causing Congenital Cataract: Case Study of a Chinese Family

Congenital cataract is the most important global cause of visual impairment in children. Autosomal dominant and autosomal recessive inheritance account for the majority of the hereditary nonsyndromic congenital cataract. The function of FYCO1 gene is to guide the transport of the microtubule-directe...

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Autores principales: Mei, Shuping, Lin, Jingwei, Liu, Zhen, Li, Cheng
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Hindawi 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9439885/
https://www.ncbi.nlm.nih.gov/pubmed/36061348
http://dx.doi.org/10.1155/2022/5838104
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author Mei, Shuping
Lin, Jingwei
Liu, Zhen
Li, Cheng
author_facet Mei, Shuping
Lin, Jingwei
Liu, Zhen
Li, Cheng
author_sort Mei, Shuping
collection PubMed
description Congenital cataract is the most important global cause of visual impairment in children. Autosomal dominant and autosomal recessive inheritance account for the majority of the hereditary nonsyndromic congenital cataract. The function of FYCO1 gene is to guide the transport of the microtubule-directed vesicles. Mutations in the FYCO1 gene may cause cataracts. We reported a novel nonsense mutation in FYCO1 (c.1411C > T, P. R471 ∗), which could cause nonsyndrome autosomal recessive congenital cataract. We underwent an ophthalmology examination of all participants and collected blood samples from all participants and extracted genomic DNAs. By whole exome sequencing, we found that this family carried an unreported mutation in the FYCO1 gene: c.1411C > T, P. R471 ∗. Sanger sequencing was performed to verify the mutation. We used ITASSER and PYMOL to predict and compare the structure and function of the mutated proteins. Using SIFT software and referring to the relevant guidelines of ACMG, the mutation was determined to be pathogenic. The models suggested that the nonsense mutation p.R471∗ resulted in a profound disruption of the FYCO1 protein structure. This report expands the locus information of the FYCO1 mutations.
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spelling pubmed-94398852022-09-03 A Novel Mutation in the FYCO1 Gene Causing Congenital Cataract: Case Study of a Chinese Family Mei, Shuping Lin, Jingwei Liu, Zhen Li, Cheng Dis Markers Research Article Congenital cataract is the most important global cause of visual impairment in children. Autosomal dominant and autosomal recessive inheritance account for the majority of the hereditary nonsyndromic congenital cataract. The function of FYCO1 gene is to guide the transport of the microtubule-directed vesicles. Mutations in the FYCO1 gene may cause cataracts. We reported a novel nonsense mutation in FYCO1 (c.1411C > T, P. R471 ∗), which could cause nonsyndrome autosomal recessive congenital cataract. We underwent an ophthalmology examination of all participants and collected blood samples from all participants and extracted genomic DNAs. By whole exome sequencing, we found that this family carried an unreported mutation in the FYCO1 gene: c.1411C > T, P. R471 ∗. Sanger sequencing was performed to verify the mutation. We used ITASSER and PYMOL to predict and compare the structure and function of the mutated proteins. Using SIFT software and referring to the relevant guidelines of ACMG, the mutation was determined to be pathogenic. The models suggested that the nonsense mutation p.R471∗ resulted in a profound disruption of the FYCO1 protein structure. This report expands the locus information of the FYCO1 mutations. Hindawi 2022-08-26 /pmc/articles/PMC9439885/ /pubmed/36061348 http://dx.doi.org/10.1155/2022/5838104 Text en Copyright © 2022 Shuping Mei et al. https://creativecommons.org/licenses/by/4.0/This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Research Article
Mei, Shuping
Lin, Jingwei
Liu, Zhen
Li, Cheng
A Novel Mutation in the FYCO1 Gene Causing Congenital Cataract: Case Study of a Chinese Family
title A Novel Mutation in the FYCO1 Gene Causing Congenital Cataract: Case Study of a Chinese Family
title_full A Novel Mutation in the FYCO1 Gene Causing Congenital Cataract: Case Study of a Chinese Family
title_fullStr A Novel Mutation in the FYCO1 Gene Causing Congenital Cataract: Case Study of a Chinese Family
title_full_unstemmed A Novel Mutation in the FYCO1 Gene Causing Congenital Cataract: Case Study of a Chinese Family
title_short A Novel Mutation in the FYCO1 Gene Causing Congenital Cataract: Case Study of a Chinese Family
title_sort novel mutation in the fyco1 gene causing congenital cataract: case study of a chinese family
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9439885/
https://www.ncbi.nlm.nih.gov/pubmed/36061348
http://dx.doi.org/10.1155/2022/5838104
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