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一种新的COL4A1基因突变导致一个中国家庭患常染色体显性非综合征性先天性白内障。

A novel COL4A1 gene mutation results in autosomal dominant non-syndromic congenital cataract in a Chinese family.

作者信息

Xia Xin-Yi, Li Na, Cao Xiang, Wu Qiu-Yue, Li Tian-Fu, Zhang Cui, Li Wei-Wei, Cui Ying-Xia, Li Xiao-Jun, Xue Chun-Yan

机构信息

Institute of Laboratory Medicine, Jinling Hospital, Nanjing University School of Medicine, 305 East Zhongshan Road, Nanjing 210002, People's Republic of China.

出版信息

BMC Med Genet. 2014 Aug 15;15:97. doi: 10.1186/s12881-014-0097-2.

Abstract

BACKGROUND

Almost one-third of congenital cataracts are primarily autosomal dominant disorders, which are also called autosomal dominant congenital cataract, resulting in blindness and clouding of the lens. The purpose of this study was to identify the disease-causing mutation in a Chinese family affected by bilateral, autosomal dominant congenital cataract.

METHODS

The detection of candidate gene mutation and the linkage analysis of microsatellite markers were performed for the known candidate genes. Molecular mapping and cloning of candidate genes were used in all affected family members to screen for potential genetic mutations and the mutation was confirmed by single enzyme digestion.

RESULTS

The proband was diagnosed with isolated, congenital cataract without the typical clinical manifestations of cataract, which include diabetes, porencephaly, sporadic intracerebral hemorrhage, and glomerulopathy. A novel mutation, c.2345 G > C (Gly782Ala), in exon 31 of the collagen type IV αlpha1 (COL4A1) gene, which encodes the collagen alpha-1(IV) chain, was found to be associated with autosomal dominant congenital cataract in a Chinese family. This mutation was not found in unaffected family members or in 200 unrelated controls. Sequence analysis confirmed that the Gly782 amino acid residue is highly conserved.

CONCLUSIONS

The novel mutation (c.2345 G > C) of the COL4A1 gene is the first report of a non-syndromic, autosomal dominant congenital cataract, thereby highlighting the important role of type IV collagen in the physiological and optical properties of the lens.

摘要

背景

几乎三分之一的先天性白内障主要是常染色体显性疾病,也被称为常染色体显性先天性白内障,可导致失明和晶状体混浊。本研究的目的是确定一个患双侧常染色体显性先天性白内障的中国家庭中的致病突变。

方法

对已知的候选基因进行候选基因突变检测和微卫星标记的连锁分析。对所有患病家庭成员进行候选基因的分子定位和克隆,以筛选潜在的基因突变,并通过单酶切确认该突变。

结果

先证者被诊断为孤立性先天性白内障,无白内障的典型临床表现,如糖尿病、脑穿通畸形、散发性脑出血和肾小球病。在编码IV型胶原α1(COL4A1)链的IV型胶原α1(COL4A1)基因第31外显子中发现一个新的突变,c.2345 G>C(Gly782Ala),该突变与一个中国家庭的常染色体显性先天性白内障相关。在未患病的家庭成员或200名无关对照中未发现该突变。序列分析证实Gly782氨基酸残基高度保守。

结论

COL4A1基因的新突变(c.2345 G>C)是关于非综合征性常染色体显性先天性白内障的首次报道,从而突出了IV型胶原在晶状体生理和光学特性中的重要作用。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6f46/4236509/b58d1610d1ec/s12881-014-0097-2-1.jpg

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