TAZ基因剪接突变导致外显子跳变和Barth综合征。

Q3 Medicine Acta Myologica Pub Date : 2021-06-30 eCollection Date: 2021-06-01 DOI:10.36185/2532-1900-047
Larysa Sivitskaya, Nina Danilenko, Iryna Motuk, Nikolai Zhelev
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引用次数: 0

摘要

Barth综合征是一种单基因x连锁疾病,以心肌病、骨骼肌病和中性粒细胞减少为特征。它是由心磷脂缺乏引起的,与他法津基因(TAZ)的突变有关。本文报告1例3岁男童,伴有扩张性心肌病、中性粒细胞减少和生长迟缓。遗传筛选发现TAZ基因2内含子和3外显子交界处有一个新的变异——c.239-1_239delinsTT。变异的功能分析显示,外显子3的异常剪接导致其在tafazzin开始时从成熟mRNA和移码上完全切除。根据在TAZ突变携带者中观察到的剪接改变和典型的临床表型,可以将c.239-1_239delinsTT变异分类为致病性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

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Splicing mutation in TAZ gene leading to exon skipping and Barth syndrome.

Barth syndrome is a monogenic X-linked disorder characterized by cardiomyopathy, skeletal myopathy and neutropenia. It is caused by deficiency of cardiolipin and associated with mutations in the tafazzin gene (TAZ). A 3 years old boy with dilated cardiomyopathy, neutropenia and growth retardation was investigated. Genetic screening found a new variant in the junction of intron 2 and exon 3 of the TAZ gene - c.239-1_239delinsTT. Functional analysis of the variant revealed the aberrant splicing of exon 3 leading to its complete excision from mature mRNA and frameshift at the beginning of tafazzin. Variant c.239-1_239delinsTT can be classified as pathogenic based on splicing alteration and typical clinical phenotype observed in TAZ mutation carriers.

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来源期刊
Acta Myologica
Acta Myologica Medicine-Cardiology and Cardiovascular Medicine
CiteScore
3.70
自引率
0.00%
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0
期刊最新文献
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