Haemostasis Research Group, Department of Cardiovascular Science, Faculty of Medicine, Dentistry and Health, University of Sheffield, Beech Hill Road, Sheffield, UK.
Thromb Haemost. 2013 Aug;110(2):264-74. doi: 10.1160/TH13-02-0135. Epub 2013 May 23.
Several cohort studies have investigated the molecular basis of von Willebrand disease (VWD); however, these have mostly focused on European and North American populations. This study aimed to investigate mutation spectrum in 26 index cases (IC) from Turkey diagnosed with all three VWD types, the majority (73%) with parents who were knowingly related. IC were screened for mutations using multiplex ligation-dependent probe amplification and analysis of all von Willebrand factor gene (VWF) exons and exon/intron boundaries. Selected missense mutations were expressed in vitro. Candidate VWF mutations were identified in 25 of 26 IC and included propeptide missense mutations in four IC (two resulting in type 1 and two in recessive 2A), all influencing VWF expression in vitro. Four missense mutations, a nonsense mutation and a small in-frame insertion resulting in type 2A were also identified. Of 15 type 3 VWD IC, 13 were homozygous and two compound heterozygous for 14 candidate mutations predicted to result in lack of expression and two propeptide missense changes. Identification of intronic breakpoints of an exon 17-18 deletion suggested that the mutation resulted from non-homologous end joining. This study provides further insight into the pathogenesis of VWD in a population with a high degree of consanguineous partnerships.
已有多项队列研究探讨了血管性血友病(VWD)的分子基础;然而,这些研究主要集中在欧洲和北美人群。本研究旨在调查 26 名经确诊患有所有三种 VWD 类型的土耳其索引病例(IC)的突变谱,其中大多数(73%)的父母为近亲。使用多重连接依赖性探针扩增和所有血管性血友病因子基因(VWF)外显子和外显子/内含子边界的分析对 IC 进行突变筛查。在体外表达了选定的错义突变。在 26 名 IC 中,有 25 名确定了候选 VWF 突变,其中包括 4 名 IC 的前肽错义突变(两种导致 1 型,两种导致隐性 2A),所有这些突变均影响体外 VWF 的表达。还发现了四个错义突变、一个无义突变和一个导致 2A 的小框内插入突变。在 15 名 3 型 VWD IC 中,有 13 名是 14 个候选突变的纯合子,有 2 名是复合杂合子,这些突变预计会导致缺乏表达和两个前肽错义改变。exon17-18 缺失的内含子断点的鉴定表明,该突变是由非同源末端连接引起的。本研究为高发近亲联姻人群的 VWD 发病机制提供了进一步的认识。