结核性硬化症外显子TSC1和TSC2基因变异的剪接分析

IF 3.3 2区 医学 Q2 GENETICS & HEREDITY
Qingqing You, Jingwei Liu, Ran Zhang, Zhi Wang, Bingying Zhang, Wencong Guo, Ning Xu, Irene Bottillo, Leping Shao
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引用次数: 0

摘要

结节性硬化症(TSC)的特点是细胞增殖和迁移异常,导致错构瘤、良性肿瘤或恶性癌症的发展,影响皮肤和大脑,以及潜在地影响心脏、肾脏、肺和眼睛,在一生中有不同的受累模式。它主要由TSC1和TSC2基因突变引起。异常剪接是遗传性疾病的重要因素。选择性剪接是扩大人类蛋白质组多样性的关键机制。破坏典型剪接位点或剪接调控元件的突变阻碍了剪接位点的利用,导致外显子跳变和内含子保留。我们利用生物信息学工具全面分析了TSC1和TSC2基因的错义和无义突变,并通过minigene分析确定了10个影响pre-mRNA剪接的候选突变。TSC基因突变可通过复杂的机制导致部分或完全外显子跳跃和/或内含子保留。本研究强调了评估它们在TSC疑似致病变异体剪接中的作用的重要性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

Splicing Analysis of Exonic TSC1 and TSC2 Gene Variants Causing Tuberous Sclerosis Complex

Splicing Analysis of Exonic TSC1 and TSC2 Gene Variants Causing Tuberous Sclerosis Complex

Tuberous sclerosis complex (TSC) is characterized by abnormalities in cell proliferation and migration, leading to the development of hamartomas, benign tumors, or malignant cancers, affecting both the skin and brain, as well as potentially impacting the heart, kidneys, lungs, and eyes, with varying patterns of involvement over a lifetime. It is primarily caused by mutations in the TSC1 and TSC2 genes. Aberrant splicing is a crucial factor in hereditary diseases. Alternative splicing is a key mechanism for expanding the diversity of the human proteome. Mutations disrupting canonical splice sites or splicing regulatory elements impede the utilization of splice sites, leading to exon skipping and intron retention. We comprehensively analyzed missense and nonsense mutations of TSC1 and TSC2 genes using bioinformatics tools and identified 10 candidate mutations affecting pre-mRNA splicing through minigene analysis. Mutations in TSC genes can lead to partial or complete exon skipping and/or intron retention through complex mechanisms. This study emphasizes the importance of evaluating their roles in the splicing of suspected pathogenic variants in TSC.

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来源期刊
Human Mutation
Human Mutation 医学-遗传学
CiteScore
8.40
自引率
5.10%
发文量
190
审稿时长
2 months
期刊介绍: Human Mutation is a peer-reviewed journal that offers publication of original Research Articles, Methods, Mutation Updates, Reviews, Database Articles, Rapid Communications, and Letters on broad aspects of mutation research in humans. Reports of novel DNA variations and their phenotypic consequences, reports of SNPs demonstrated as valuable for genomic analysis, descriptions of new molecular detection methods, and novel approaches to clinical diagnosis are welcomed. Novel reports of gene organization at the genomic level, reported in the context of mutation investigation, may be considered. The journal provides a unique forum for the exchange of ideas, methods, and applications of interest to molecular, human, and medical geneticists in academic, industrial, and clinical research settings worldwide.
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