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全外显子组测序在一个大型的多效家族中鉴定出 TP53BP2 为原发性开角型青光眼的新候选基因。

Identification of TP53BP2 as a Novel Candidate Gene for Primary Open Angle Glaucoma by Whole Exome Sequencing in a Large Multiplex Family.

机构信息

Department of Ophthalmology, Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center, Nijmegen, the Netherlands.

Department of Clinical Genetics, Academic Medical Centre, Amsterdam, the Netherlands.

出版信息

Mol Neurobiol. 2018 Feb;55(2):1387-1395. doi: 10.1007/s12035-017-0403-z. Epub 2017 Feb 1.

Abstract

Primary open angle glaucoma (POAG) is a major type of glaucoma characterized by progressive loss of retinal ganglion cells with associated visual field loss without an identifiable secondary cause. Genetic factors are considered to be major contributors to the pathogenesis of glaucoma. The aim of the study was to identify the causative gene in a large family with POAG by applying whole exome sequencing (WES). WES was performed on the DNA of four affected family members. Rare pathogenic variants shared among the affected individuals were filtered. Polymerase chain reaction and Sanger sequencing were used to analyze variants segregating with the disease in additional family members. WES analysis identified a variant in TP53BP2 (c.109G>A; p.Val37Met) that segregated heterozygously with the disease. In silico analysis of the substitution predicted it to be pathogenic. The variant was absent in public databases and in 180 population-matched controls. A novel genetic variant in the TP53BP2 gene was identified in a family with POAG. Interestingly, it has previously been demonstrated that the gene regulates apoptosis in retinal ganglion cells. This supports that the TP53BP2 variant may represent the cause of POAG in this family. Additional screening of the gene in patients with POAG from different populations is required to confirm its involvement in the disease.

摘要

原发性开角型青光眼(POAG)是一种主要的青光眼类型,其特征是视网膜神经节细胞进行性丧失,并伴有视野丧失,而没有可识别的继发原因。遗传因素被认为是青光眼发病机制的主要因素。本研究旨在通过全外显子组测序(WES)鉴定一个具有 POAG 的大型家族的致病基因。对 4 名受影响家庭成员的 DNA 进行了 WES。筛选出受影响个体之间共享的罕见致病性变异。聚合酶链反应和 Sanger 测序用于分析与疾病共分离的变体在其他家族成员中的情况。WES 分析鉴定了 TP53BP2 基因中的一个变体(c.109G>A;p.Val37Met),该变体与疾病呈杂合状态共分离。替代物的计算机分析预测其具有致病性。该变体不存在于公共数据库和 180 个人群匹配对照中。在具有 POAG 的家族中鉴定出了 TP53BP2 基因中的一个新的遗传变异。有趣的是,先前已经证明该基因调节视网膜神经节细胞的细胞凋亡。这支持该 TP53BP2 变体可能是该家族 POAG 的原因。需要对来自不同人群的 POAG 患者进行该基因的进一步筛选,以确认其与该疾病的相关性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b1b8/5820370/6c8ad42e600b/12035_2017_403_Fig1_HTML.jpg

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