Literature DB >> 11057846

Molecular characterization of a multiethnic group of 21 patients with type 3 von Willebrand disease.

L Baronciani1, G Cozzi, M T Canciani, F Peyvandi, A Srivastava, A B Federici, P M Mannucci.   

Abstract

Type 3 von Willebrand disease is a rare autosomal disorder characterized by unmeasurable levels of von Willebrand factor and severe hemorrhagic symptoms. We studied a multiethnic group of 37 patients, from Italy (n = 14), Iran (n = 10) and India(n = 13) to identify the molecular defects and to evaluate genetic heterogeneity among these populations. Twenty-one patients (6 Italians, 9 Iranians and 6 Indians) were fully characterized at the molecular level. Twenty-four different gene alterations were identified, 20 of which have not been described previously. The majority of the mutations caused null alleles, 11 being nonsense mutations (Q218*, W222*, R365*, R373*, E644*, Q706*, S1338*, Q1346*, Y1542*, R1659*, E2129*), 4 small deletions (437delG, 2680delC, 6431delT, del 8491-8499), 3 possible splice site mutations [IVS9(-1)g-->a, IVS29(+10)c-->t, IVS40(-1)g-->c], 3 candidate missense mutations (C275S, C2174G, C2804Y), 2 small insertions (7375insC, 7921insC) and 1 large gene deletion. The latter mutation was associated with the development of alloantibodies to VWF, but this complication was also found in a patient homozygous for a nonsense mutation (Q1346*). Due to the ethnic origin of the patients most of them were the offspring of consanguineous marriages and so were homozygous for the mutations found (18/21). Our results indicate that molecular defects responsible for type 3 VWD are scattered throughout the entire VWF gene (from exon 3 to 52), and that there is no prevalent and common gene defect in the three populations studied by us.

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Year:  2000        PMID: 11057846

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


  6 in total

1.  Clinical and laboratory versus molecular markers for a correct classification of von Willebrand disease.

Authors:  Augusto B Federici; Maria T Canciani
Journal:  Haematologica       Date:  2009-05       Impact factor: 9.941

2.  Pharmacokinetics and safety of a novel recombinant human von Willebrand factor manufactured with a plasma-free method: a prospective clinical trial.

Authors:  Pier Mannuccio Mannucci; Christine Kempton; Carolyn Millar; Edward Romond; Amy Shapiro; Ingvild Birschmann; Margaret V Ragni; Joan Cox Gill; Thynn Thynn Yee; Robert Klamroth; Wing-Yen Wong; Miranda Chapman; Werner Engl; Peter L Turecek; Tobias M Suiter; Bruce M Ewenstein
Journal:  Blood       Date:  2013-06-18       Impact factor: 22.113

3.  Genetic heterogeneity in a large cohort of Indian type 3 von Willebrand disease patients.

Authors:  Priyanka Kasatkar; Shrimati Shetty; Kanjaksha Ghosh
Journal:  PLoS One       Date:  2014-03-27       Impact factor: 3.240

4.  Genotypes of European and Iranian patients with type 3 von Willebrand disease enrolled in 3WINTERS-IPS.

Authors:  Luciano Baronciani; Ian Peake; Reinhard Schneppenheim; Anne Goodeve; Minoo Ahmadinejad; Zahra Badiee; Mohammad-Reza Baghaipour; Olga Benitez; Imre Bodó; Ulrich Budde; Andrea Cairo; Giancarlo Castaman; Peyman Eshghi; Jenny Goudemand; Wolf Hassenpflug; Hamid Hoorfar; Mehran Karimi; Bijan Keikhaei; Riitta Lassila; Frank W G Leebeek; Maria Fernanda Lopez Fernandez; Pier Mannuccio Mannucci; Renato Marino; Nikolas Nikšić; Florian Oyen; Cristina Santoro; Andreas Tiede; Gholamreza Toogeh; Alberto Tosetto; Marc Trossaert; Eva M K Zetterberg; Jeroen Eikenboom; Augusto B Federici; Flora Peyvandi
Journal:  Blood Adv       Date:  2021-08-10

5.  The role of exon 45 and 16 in the pathogenesis of Von Willebrand disease in Iranian Patients.

Authors:  M Nasiri; H Galehdari; M Darbouy; M Yavarian; B Keikhaee
Journal:  Iran J Ped Hematol Oncol       Date:  2012-09-22

6.  Additional markers for genetic diagnosis of type 3 von Willebrand disease in Indian population.

Authors:  Priyanka Kasatkar; Kanjaksha Ghosh; Shrimati Shetty
Journal:  Indian J Med Res       Date:  2015-12       Impact factor: 2.375

  6 in total

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