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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.
- 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.
- Subjects :
- Adult
Base Sequence
Child
Child, Preschool
Codon, Nonsense genetics
Frameshift Mutation genetics
Genes, Recessive genetics
Genotype
Humans
Infant
Male
Molecular Sequence Data
Phenotype
Polymerase Chain Reaction
Polymorphism, Single-Stranded Conformational
Proteins chemistry
Sequence Homology, Nucleic Acid
Wiskott-Aldrich Syndrome Protein
Mutation genetics
Proteins genetics
Wiskott-Aldrich Syndrome genetics
Subjects
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