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CRYGC 中的新突变与中国家族性先天性白内障有关。

Novel mutations in CRYGC are associated with congenital cataracts in Chinese families.

机构信息

Department of Ophthalmology of Shanghai Tenth People's Hospital, and Tongji Eye Institute, Tongji University School of Medicine, Shanghai, China.

Department of Medical Genetics, Tongji University School of Medicine, Shanghai, China.

出版信息

Sci Rep. 2017 Mar 15;7(1):189. doi: 10.1038/s41598-017-00318-1.

Abstract

Congenital cataract (CC), responsible for about one-third of blindness in infants, is a major cause of vision loss in children worldwide. 10-25% of CC cases are attributed to genetic causes and CC is a clinically and genetically highly heterogeneous lens disorder in children. Autosomal dominant (AD) inheritance is the most commonly pattern. 195 unrelated non-syndromic ADCC families in this study are recruited from 15 provinces of China. Sanger sequencing approach followed by intra-familial co-segregation, in Silico analyses and interpretation of the variations according to the published guidelines of American College of Medical Genetics (ACMG), were employed to determine the genetic defects. Two mutations (p.Tyr139X and p.Ser166Phe) identified in two unrelated families were associated with their congenital nuclear cataracts and microcornea respectively, which are also reported previously. Six novel CRYGC mutations (p.Asp65ThrfsX38, p.Arg142GlyfsX5, p.Arg142AlafsX22, p.Tyr144X, p.Arg169X, and p.Tyr46Asp) were identified in other six families with congenital nuclear cataracts, respectively. Mutations in the CRYGC were responsible for 4.1% Chinese ADCC families in our cohort. Our results expand the spectrum of CRYGC mutations as well as their associated phenotypes.

摘要

先天性白内障(CC)约占婴儿失明的三分之一,是全球儿童视力丧失的主要原因。10-25%的 CC 病例归因于遗传原因,CC 是儿童中一种临床和遗传上高度异质的晶状体疾病。常染色体显性(AD)遗传是最常见的模式。本研究从中国 15 个省招募了 195 个无综合征的 ADCC 家族,每个家族都有 195 个无血缘关系的非综合征性 ADCC 家庭。采用桑格测序方法,然后进行家系内共分离、计算机分析和根据美国医学遗传学学院(ACMG)发布的指南对变异进行解释,以确定遗传缺陷。在两个无血缘关系的家庭中发现的两种突变(p.Tyr139X 和 p.Ser166Phe)分别与他们的先天性核白内障和小角膜有关,这两种突变之前也有报道。在其他 6 个先天性核白内障的家庭中分别发现了 6 个新的 CRYGC 突变(p.Asp65ThrfsX38、p.Arg142GlyfsX5、p.Arg142AlafsX22、p.Tyr144X、p.Arg169X 和 p.Tyr46Asp)。CRYGC 突变导致我们队列中 4.1%的中国 ADCC 家庭。我们的结果扩展了 CRYGC 突变及其相关表型的范围。

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