Literature DB >> 9198195

Gene defects in 150 unrelated French cases with type 2 von Willebrand disease: from the patient to the gene. INSERM Network on Molecular Abnormalities in von Willebrand Disease.

D Meyer1, E Fressinaud, C Gaucher, J M Lavergne, L Hilbert, A S Ribba, S Jorieux, C Mazurier.   

Abstract

Type 2 vWD is defined by qualitative defects of vWF and is subdivided into four subtypes: 2N, 2B, 2A and 2M. The characterization of 150 unrelated French cases with type 2 vWD emphasizes the heterogeneity of this group. In 51 cases of type 2N vWD, new mutations were found not only in the D' domain (Cys25Tyr and Cys95Phe) but also in the D3 domain (Asp116Asn and Cys297Arg). In 42 cases of type 2B vWD, no new mutation was detected. In 45 cases with type 2A phenotype, three new candidate mutations were found in the A2 domain: Gln793Arg, Val841Phe and Leu876Pro. In addition, four new candidate mutations were detected in the A1 domain: Cys509Gly, Arg545His, Arg552Cys and Cys695Tyr. Finally, five new candidate mutations were identified in 12 patients with 2M (or unclassified) phenotype: Leu513Pro, Gly561A1a, Glu596Lys, Arg611Leu and IIe662Phe. For all candidate mutations, expression studies are in progress. This study of a large number of French variants of vWD brings further insight into the relationship between phenotype and genotype.

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Year:  1997        PMID: 9198195

Source DB:  PubMed          Journal:  Thromb Haemost        ISSN: 0340-6245            Impact factor:   5.249


  12 in total

1.  Selectin-like kinetics and biomechanics promote rapid platelet adhesion in flow: the GPIb(alpha)-vWF tether bond.

Authors:  Teresa A Doggett; Gaurav Girdhar; Avril Lawshé; David W Schmidtke; Ian J Laurenzi; Scott L Diamond; Thomas G Diacovo
Journal:  Biophys J       Date:  2002-07       Impact factor: 4.033

2.  Molecular coevolution of coagulation factor VIII and von Willebrand factor.

Authors:  Philip M Zakas; Christopher W Coyle; Anja Brehm; Marion Bayer; Barbara Solecka-Witulska; Caelan E Radford; Christine Brown; Kate Nesbitt; Courtney Dwyer; Christoph Kannicht; H Trent Spencer; Eric A Gaucher; Christopher B Doering; David Lillicrap
Journal:  Blood Adv       Date:  2021-02-09

3.  A common ancestor more than 10,000 years old for patients with R854Q-related type 2N von Willebrand's disease in Italy.

Authors:  Alessandra Casonato; Viviana Daidone; Giovanni Barbon; Elena Pontara; Irene Di Pasquale; Lisa Gallinaro; Letizia Marullo; Giorgio Bertorelle
Journal:  Haematologica       Date:  2012-08-08       Impact factor: 9.941

Review 4.  von Willebrand disease: clinical and laboratory lessons learned from the large von Willebrand disease studies.

Authors:  Paula D James; David Lillicrap
Journal:  Am J Hematol       Date:  2012-03-03       Impact factor: 10.047

Review 5.  Molecular genetics of type 2 von Willebrand disease.

Authors:  Edith Fressinaud; Claudine Mazurier; Dominique Meyer
Journal:  Int J Hematol       Date:  2002-01       Impact factor: 2.490

6.  Novel insights into the clinical phenotype and pathophysiology underlying low VWF levels.

Authors:  Michelle Lavin; Sonia Aguila; Sonja Schneppenheim; Niall Dalton; Kenneth L Jones; Jamie M O'Sullivan; Niamh M O'Connell; Kevin Ryan; Barry White; Mary Byrne; Marie Rafferty; Mairead M Doyle; Margaret Nolan; Roger J S Preston; Ulrich Budde; Paula James; Jorge Di Paola; James S O'Donnell
Journal:  Blood       Date:  2017-09-15       Impact factor: 22.113

7.  Variability in platelet- and collagen-binding defects in type 2M von Willebrand disease.

Authors:  D M Larsen; S L Haberichter; J C Gill; A D Shapiro; V H Flood
Journal:  Haemophilia       Date:  2013-03-18       Impact factor: 4.287

8.  Extensive contacts between ADAMTS13 exosites and von Willebrand factor domain A2 contribute to substrate specificity.

Authors:  Weiqiang Gao; Patricia J Anderson; J Evan Sadler
Journal:  Blood       Date:  2008-05-20       Impact factor: 22.113

Review 9.  The molecular characterization of von Willebrand disease: good in parts.

Authors:  P D James; D Lillicrap
Journal:  Br J Haematol       Date:  2013-02-14       Impact factor: 6.998

10.  A molten globule intermediate of the von Willebrand factor A1 domain firmly tethers platelets under shear flow.

Authors:  Alexander Tischer; Pranathi Madde; Luis M Blancas-Mejia; Matthew Auton
Journal:  Proteins       Date:  2013-11-22
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