Literature DB >> 33497541

Von Willebrand disease type 2N: An update.

Omid Seidizadeh1, Flora Peyvandi1,2, Pier Mannuccio Mannucci1.   

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.
© 2021 International Society on Thrombosis and Haemostasis.

Entities:  

Keywords:  factor VIII; factor VIII binding; gene mutation; von Willebrand factor

Mesh:

Substances:

Year:  2021        PMID: 33497541     DOI: 10.1111/jth.15247

Source DB:  PubMed          Journal:  J Thromb Haemost        ISSN: 1538-7836            Impact factor:   5.824


  5 in total

1.  Cryptogenic oozers and bruisers.

Authors:  Kristi J Smock; Karen A Moser
Journal:  Hematology Am Soc Hematol Educ Program       Date:  2021-12-10

2.  Phenotypic and genetic characterizations of the Milan cohort of von Willebrand disease type 2.

Authors:  Omid Seidizadeh; Luciano Baronciani; Maria Teresa Pagliari; Giovanna Cozzi; Paola Colpani; Andrea Cairo; Simona Maria Siboni; Eugenia Biguzzi; Flora Peyvandi
Journal:  Blood Adv       Date:  2022-07-12

Review 3.  Prophylactic management of patients with von Willebrand disease.

Authors:  Massimo Franchini; Omid Seidizadeh; Pier Mannuccio Mannucci
Journal:  Ther Adv Hematol       Date:  2021-12-22

4.  A novel mouse model of type 2N VWD was developed by CRISPR/Cas9 gene editing and recapitulates human type 2N VWD.

Authors:  Qizhen Shi; Scot A Fahs; Jeremy G Mattson; Hongyin Yu; Crystal L Perry; Patricia A Morateck; Jocelyn A Schroeder; Jessica Rapten; Hartmut Weiler; Robert R Montgomery
Journal:  Blood Adv       Date:  2022-05-10

Review 5.  The Vascular Endothelium and Coagulation: Homeostasis, Disease, and Treatment, with a Focus on the Von Willebrand Factor and Factors VIII and V.

Authors:  Juan A De Pablo-Moreno; Luis Javier Serrano; Luis Revuelta; María José Sánchez; Antonio Liras
Journal:  Int J Mol Sci       Date:  2022-07-27       Impact factor: 6.208

  5 in total

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