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2N型血管性血友病:最新进展

Von Willebrand disease type 2N: An update.

作者信息

Seidizadeh Omid, Peyvandi Flora, Mannucci Pier Mannuccio

机构信息

Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Angelo Bianchi Bonomi Hemophilia and Thrombosis Center and Fondazione Luigi Villa, Milan, Italy.

Department of Pathophysiology and Transplantation, Università degli Studi di Milano, Milan, Italy.

出版信息

J Thromb Haemost. 2021 Apr;19(4):909-916. doi: 10.1111/jth.15247. Epub 2021 Mar 26.

DOI:10.1111/jth.15247
PMID:33497541
Abstract

Quantitative or qualitative defects of von Willebrand factor (VWF) are responsible for the most common inherited bleeding disorder, von Willebrand disease (VWD). Type 2N VWD is an uncommon recessive disorder that results from gene mutations located in the region coding for the binding site of VWF for factor VIII (FVIII). This narrative review describes the pathophysiology, diagnostic procedures and treatment as well as the molecular biology of type 2N VWD. Although other VWF-dependent functions like binding to platelets and collagen are preserved, FVIII plasma levels are low due to the rapid clearance of this moiety in the absence or reduction of its binding to VWF. The diagnosis of type 2N should be considered in patients with low FVIII coagulant activity (FVIII:C) and disproportionally higher VWF antigen, especially when they present with an autosomal recessive pattern of inheritance. Because an accurate diagnosis is essential for genetic counseling and optimal treatment, type 2N must be distinguished from mild/moderate hemophilia A and its carrier state. This differential diagnosis can be obtained by using the laboratory assay of the FVIII binding capacity of VWF (VWF:FVIIIB) or analysis of the FVIII binding site on the VWF gene.

摘要

血管性血友病因子(VWF)的定量或定性缺陷是导致最常见的遗传性出血性疾病——血管性血友病(VWD)的原因。2N型VWD是一种罕见的隐性疾病,由位于VWF与凝血因子VIII(FVIII)结合位点编码区域的基因突变引起。这篇叙述性综述描述了2N型VWD的病理生理学、诊断程序、治疗以及分子生物学。尽管其他VWF依赖性功能(如与血小板和胶原蛋白的结合)得以保留,但由于FVIII在缺乏或减少与VWF结合时的快速清除,FVIII血浆水平较低。对于FVIII凝血活性(FVIII:C)低且VWF抗原不成比例地升高的患者,尤其是呈现常染色体隐性遗传模式的患者,应考虑2N型的诊断。由于准确诊断对于遗传咨询和最佳治疗至关重要,2N型必须与轻度/中度A型血友病及其携带者状态相区分。这种鉴别诊断可以通过使用VWF的FVIII结合能力实验室检测(VWF:FVIIIB)或分析VWF基因上的FVIII结合位点来实现。

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