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包含151个基因的卷曲螺旋结构域(CCDC151)中的无义突变导致原发性纤毛运动障碍。

Nonsense mutation in coiled-coil domain containing 151 gene (CCDC151) causes primary ciliary dyskinesia.

作者信息

Alsaadi Muslim M, Erzurumluoglu A Mesut, Rodriguez Santiago, Guthrie Philip A I, Gaunt Tom R, Omar Hager Z, Mubarak Mohammad, Alharbi Khalid K, Al-Rikabi Ammar C, Day Ian N M

机构信息

College of Medicine, King Saud University, Riyadh, 11533, Kingdom of Saudi Arabia.

出版信息

Hum Mutat. 2014 Dec;35(12):1446-8. doi: 10.1002/humu.22698.

Abstract

Primary ciliary dyskinesia (PCD) is an autosomal-recessive disorder characterized by impaired ciliary function that leads to subsequent clinical phenotypes such as chronic sinopulmonary disease. PCD is also a genetically heterogeneous disorder with many single gene mutations leading to similar clinical phenotypes. Here, we present a novel PCD causal gene, coiled-coil domain containing 151 (CCDC151), which has been shown to be essential in motile cilia of many animals and other vertebrates but its effects in humans was not observed until currently. We observed a novel nonsense mutation in a homozygous state in the CCDC151 gene (NM_145045.4:c.925G>T:p.[E309*]) in a clinically diagnosed PCD patient from a consanguineous family of Arabic ancestry. The variant was absent in 238 randomly selected individuals indicating that the variant is rare and likely not to be a founder mutation. Our finding also shows that given prior knowledge from model organisms, even a single whole-exome sequence can be sufficient to discover a novel causal gene.

摘要

原发性纤毛运动障碍(PCD)是一种常染色体隐性疾病,其特征是纤毛功能受损,进而导致慢性鼻窦肺疾病等临床表型。PCD也是一种基因异质性疾病,许多单基因突变会导致相似的临床表型。在此,我们报告了一个新的PCD致病基因,即卷曲螺旋结构域包含蛋白151(CCDC151),该基因在许多动物和其他脊椎动物的运动纤毛中已被证明是必需的,但直到目前才观察到其在人类中的作用。我们在一名来自阿拉伯血统近亲家庭的临床诊断PCD患者中,观察到CCDC151基因(NM_145045.4:c.925G>T:p.[E309*])存在纯合状态的新型无义突变。在238名随机选择的个体中未发现该变异,表明该变异罕见,且可能不是奠基者突变。我们的研究结果还表明,鉴于模式生物的先验知识,即使是单个全外显子测序也足以发现一个新的致病基因。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2c16/4489323/6f63415e73bc/humu0035-1446-f1.jpg

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