Literature DB >> 8682510

Wiskott-Aldrich syndrome: no strict genotype-phenotype correlations but clustering of missense mutations in the amino-terminal part of the WASP gene product.

D Schindelhauer1, M Weiss, H Hellebrand, A Golla, M Hergersberg, R Seger, B H Belohradsky, A Meindl.   

Abstract

The Wiskott-Aldrich syndrome protein (WASP) gene was found to be mutated in patients presenting with WAS and in patients showing X-linked thrombocytopenia. Mutation analysis in 19 families of German, Swiss and Turkish descent by single-strand conformation polymorphism and sequencing resulted in the detection of seven novel and 10 known mutations. A striking clustering of missense mutations in the first four exons contrasted with a random distribution of nonsense mutations. More than 85% of all known missense mutations were localized in the amino-terminal stretch of the WASP gene product; this region contained a mutational hot spot at codon 86. No genotype-phenotype correlation emerged after a comparison of the identified mutations with the resulting clinical picture for a classical WAS phenotype. A substitution at codon 86 resulted in an extremely variable expression of the disease in a large Swiss family. An extended homology search revealed a distant relationship of this stretch to the vasodilator-stimulated phosphoprotein (VASP), which is involved in the maintenance of cyto-architecture by interacting with actin-like filaments.

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Year:  1996        PMID: 8682510     DOI: 10.1007/s004390050162

Source DB:  PubMed          Journal:  Hum Genet        ISSN: 0340-6717            Impact factor:   4.132


  18 in total

Review 1.  The Wiskott-Aldrich syndrome.

Authors:  H D Ochs
Journal:  Clin Rev Allergy Immunol       Date:  2001-02       Impact factor: 8.667

Review 2.  The Wiskott-Aldrich syndrome.

Authors:  A J Thrasher; C Kinnon
Journal:  Clin Exp Immunol       Date:  2000-04       Impact factor: 4.330

3.  Mutational analysis of the WASP gene in 2 Korean families with Wiskott-Aldrich syndrome.

Authors:  Eun-Kyeong Jo; Takeshi Futatani; Hirokazu Kanegane; Takeo Kubota; Young-Ho Lee; Jin-A Jung; Chang-Hwa Song; Jeong-Kyu Park; Shigeaki Nonoyama; Toshio Miyawaki
Journal:  Int J Hematol       Date:  2003-07       Impact factor: 2.490

4.  A novel Wiskott-Aldrich syndrome protein (WASP) complex mutation identified in a WAS patient results in an aberrant product at the C-terminus from two transcripts with unusual polyA signals.

Authors:  Nuria Andreu; Maricruz García-Rodríguez; Victor Volpini; Cecilia Frecha; Ignacio J Molina; Gumersindo Fontan; Cristina Fillat
Journal:  J Hum Genet       Date:  2005-12-22       Impact factor: 3.172

Review 5.  The Wiskott-Aldrich syndrome.

Authors:  H D Ochs
Journal:  Springer Semin Immunopathol       Date:  1998

6.  WASp verprolin homology, cofilin homology, and acidic region domain-mediated actin polymerization is required for T cell development.

Authors:  Jinyi Zhang; Fabio Shi; Karen Badour; Yupu Deng; Mary K H McGavin; Katherine A Siminovitch
Journal:  Proc Natl Acad Sci U S A       Date:  2002-02-12       Impact factor: 11.205

7.  Wiskott-Aldrich syndrome protein regulates podosomes in primary human macrophages.

Authors:  S Linder; D Nelson; M Weiss; M Aepfelbacher
Journal:  Proc Natl Acad Sci U S A       Date:  1999-08-17       Impact factor: 11.205

8.  Clinical aspects and genetic analysis of taiwanese patients with wiskott-Aldrich syndrome protein mutation: the first identification of x-linked thrombocytopenia in the chinese with novel mutations.

Authors:  Wen-I Lee; Jing-Long Huang; Tang-Her Jaing; Kang-Hsi Wu; Yin-Hsiu Chien; Kuei-Wen Chang
Journal:  J Clin Immunol       Date:  2010-03-16       Impact factor: 8.317

9.  Monozygotic twin pair showing discordant phenotype for X-linked thrombocytopenia and Wiskott-Aldrich syndrome: a role for epigenetics?

Authors:  David Buchbinder; Kari Nadeau; Diane Nugent
Journal:  J Clin Immunol       Date:  2011-06-28       Impact factor: 8.317

10.  Identification and characterization of a novel splice-site mutation in a patient with Wiskott-Aldrich syndrome.

Authors:  Nuria Andreu; Carmen Carreras; Félix Prieto; Xavier Estivill; Victor Volpini; Cristina Fillat
Journal:  J Hum Genet       Date:  2003-10-18       Impact factor: 3.172

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