家族性卡瓦哈尔综合征的新型桥蛋白突变。

Q3 Medicine
Acta Myologica Pub Date : 2018-12-01
Danat Yermakovich, Larysa Sivitskaya, Tatiyana Vaikhanskaya, Nina Danilenko, Iryna Motuk
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引用次数: 0

摘要

Desmoplakin由DSP基因编码,其功能改变导致皮肤和毛发异常以及心脏病。卡瓦哈尔综合征(Carvajal syndrome, CS)具有以上三种特征。CS是一种常染色体隐性遗传病,定位于6q24染色体,由DSP基因突变引起。我们报告一位由两种新型DSP基因突变引起的CS患者,遗传自其父母,均无症状。同样的表型出现在他的妹妹身上,她表现出皮肤异常和毛茸茸的毛发。通过基因检测对已知的DSP基因位点进行分离分析,确定了患者及其妹妹的两个突变位点(C . 6986t > C和C . 7123g > C)的转位。第一个突变遗传自母亲,另一个遗传自父亲。在兄弟姐妹中产生的复合杂合突变可能是导致疾病的原因。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

Novel desmoplakin mutations in familial Carvajal syndrome.

Novel desmoplakin mutations in familial Carvajal syndrome.

Novel desmoplakin mutations in familial Carvajal syndrome.

Desmoplakin is encoded by DSP gene, whose altered function leads to skin and hair abnormalities, and heart diseases. The whole triad of these traits characterizes the Carvajal syndrome (CS). CS is an autosomal recessive genetic disorder, mapping on chromosome 6q24 and caused by mutations in DSP gene. We report a patient with CS caused by two novel mutations in DSP gene, inherited from his parents, both asymptomatic. The same phenotype was present in his younger sister who showed skin abnormality and woolly hairs. The segregation analysis of the known loci in DSP gene performed by genetic testing, was able to established the trans position of the two mutations (c.6986T > C and c.7123G > C) in the patient and his sister. The first mutation has been inherited from the mother, the other one from the father. The resulting compound heterozygous mutation in the siblings, is likely the cause of the disease.

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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
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