Larysa Sivitskaya, Nina Danilenko, Iryna Motuk, Nikolai Zhelev
{"title":"TAZ基因剪接突变导致外显子跳变和Barth综合征。","authors":"Larysa Sivitskaya, Nina Danilenko, Iryna Motuk, Nikolai Zhelev","doi":"10.36185/2532-1900-047","DOIUrl":null,"url":null,"abstract":"<p><p>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 (<i>TAZ</i>). 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.</p>","PeriodicalId":35953,"journal":{"name":"Acta Myologica","volume":"40 2","pages":"88-92"},"PeriodicalIF":0.0000,"publicationDate":"2021-06-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8290511/pdf/am-2021-02-88.pdf","citationCount":"0","resultStr":"{\"title\":\"Splicing mutation in <i>TAZ</i> gene leading to exon skipping and Barth syndrome.\",\"authors\":\"Larysa Sivitskaya, Nina Danilenko, Iryna Motuk, Nikolai Zhelev\",\"doi\":\"10.36185/2532-1900-047\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>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 (<i>TAZ</i>). 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.</p>\",\"PeriodicalId\":35953,\"journal\":{\"name\":\"Acta Myologica\",\"volume\":\"40 2\",\"pages\":\"88-92\"},\"PeriodicalIF\":0.0000,\"publicationDate\":\"2021-06-30\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8290511/pdf/am-2021-02-88.pdf\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Acta Myologica\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.36185/2532-1900-047\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2021/6/1 0:00:00\",\"PubModel\":\"eCollection\",\"JCR\":\"Q3\",\"JCRName\":\"Medicine\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Acta Myologica","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.36185/2532-1900-047","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2021/6/1 0:00:00","PubModel":"eCollection","JCR":"Q3","JCRName":"Medicine","Score":null,"Total":0}
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.