Suppr超能文献

鉴定和分析与一名土耳其患者群体相关的血管性血友病相关突变。

Identification and characterisation of mutations associated with von Willebrand disease in a Turkish patient cohort.

机构信息

Haemostasis Research Group, Department of Cardiovascular Science, Faculty of Medicine, Dentistry and Health, University of Sheffield, Beech Hill Road, Sheffield, UK.

出版信息

Thromb Haemost. 2013 Aug;110(2):264-74. doi: 10.1160/TH13-02-0135. Epub 2013 May 23.

Abstract

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.

摘要

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。

相似文献

1
Identification and characterisation of mutations associated with von Willebrand disease in a Turkish patient cohort.
Thromb Haemost. 2013 Aug;110(2):264-74. doi: 10.1160/TH13-02-0135. Epub 2013 May 23.
5
Phenotypic and molecular characterisation of type 3 von Willebrand disease in a cohort of Indian patients.
Thromb Haemost. 2013 Apr;109(4):652-60. doi: 10.1160/TH12-10-0737. Epub 2013 Feb 14.
6
The genetic basis of von Willebrand disease.
Blood Rev. 2010 May;24(3):123-34. doi: 10.1016/j.blre.2010.03.003. Epub 2010 Apr 20.
9
Laboratory diagnosis and molecular classification of von Willebrand disease.
Acta Haematol. 2009;121(2-3):71-84. doi: 10.1159/000214846. Epub 2009 Jun 8.

引用本文的文献

1
Genotypes of European and Iranian patients with type 3 von Willebrand disease enrolled in 3WINTERS-IPS.
Blood Adv. 2021 Aug 10;5(15):2987-3001. doi: 10.1182/bloodadvances.2020003397.
3
ASH ISTH NHF WFH 2021 guidelines on the diagnosis of von Willebrand disease.
Blood Adv. 2021 Jan 12;5(1):280-300. doi: 10.1182/bloodadvances.2020003265.
4
Characterization of large in-frame von Willebrand factor deletions highlights differing pathogenic mechanisms.
Blood Adv. 2020 Jul 14;4(13):2979-2990. doi: 10.1182/bloodadvances.2018027813.
6
p.P2063S: a neutral VWF variant masquerading as a mutation.
Ann Hematol. 2014 Mar;93(3):505-6. doi: 10.1007/s00277-013-1817-y. Epub 2013 Jun 18.

本文引用的文献

1
VWF propeptide and ratios between VWF, VWF propeptide, and FVIII in the characterization of type 1 von Willebrand disease.
Blood. 2013 Mar 21;121(12):2336-9. doi: 10.1182/blood-2012-09-455089. Epub 2013 Jan 24.
2
An integrated map of genetic variation from 1,092 human genomes.
Nature. 2012 Nov 1;491(7422):56-65. doi: 10.1038/nature11632.
4
VWF mutations and new sequence variations identified in healthy controls are more frequent in the African-American population.
Blood. 2012 Mar 1;119(9):2135-40. doi: 10.1182/blood-2011-10-384610. Epub 2011 Dec 23.
5
Deep intronic variations may cause mild hemophilia A.
J Thromb Haemost. 2011 Aug;9(8):1541-8. doi: 10.1111/j.1538-7836.2011.04408.x.
6
The molecular basis of von Willebrand disease: the under investigated, the unexpected and the overlooked.
Haematologica. 2011 Jun;96(6):798-800. doi: 10.3324/haematol.2011.046623.
8
Characterization of duplication breakpoints in the factor VIII gene.
J Thromb Haemost. 2010 Dec;8(12):2696-704. doi: 10.1111/j.1538-7836.2010.04040.x.
9
The genetic basis of von Willebrand disease.
Blood Rev. 2010 May;24(3):123-34. doi: 10.1016/j.blre.2010.03.003. Epub 2010 Apr 20.
10
A method and server for predicting damaging missense mutations.
Nat Methods. 2010 Apr;7(4):248-9. doi: 10.1038/nmeth0410-248.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验