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

Authors :
Schindelhauer D
Weiss M
Hellebrand H
Golla A
Hergersberg M
Seger R
Belohradsky BH
Meindl A
Source :
Human genetics [Hum Genet] 1996 Jul; Vol. 98 (1), pp. 68-76.
Publication Year :
1996

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.

Details

Language :
English
ISSN :
0340-6717
Volume :
98
Issue :
1
Database :
MEDLINE
Journal :
Human genetics
Publication Type :
Academic Journal
Accession number :
8682510
Full Text :
https://doi.org/10.1007/s004390050162