| Literature DB >> 15284122 |
Yinzhu Jin1, Cinzia Mazza, Jacinda R Christie, Silvia Giliani, Maurilia Fiorini, Patrizia Mella, Francesca Gandellini, Donn M Stewart, Qili Zhu, David L Nelson, Luigi D Notarangelo, Hans D Ochs.
Abstract
The Wiskott-Aldrich syndrome (WAS) is an X-linked recessive immune deficiency disorder characterized by thrombocytopenia, small platelet size, eczema, recurrent infections, and increased risk of autoimmune disorders and malignancies. X-linked thrombocytopenia (XLT) is an allelic variant of WAS which presents with a milder phenotype, generally limited to thrombocytopenia. WAS and XLT are caused by mutations of the Wiskott-Aldrich syndrome protein (WASP) gene which encodes a 502-amino acid protein, named WASP. WASP is thought to play a role in actin cytoskeleton organization and cell signaling. Here, we report the identification of 141 unique mutations, 71 not previously reported, from 227 WAS/XLT families with a total of 262 affected members. When possible we studied the effects of these mutations on transcription, RNA splicing, and protein expression. By analyzing a large number of patients with WAS/XLT at the molecular level we identified 5 mutational hotspots in the WASP gene and have been able to establish a strong association between genotype and phenotype.Entities:
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Year: 2004 PMID: 15284122 DOI: 10.1182/blood-2003-05-1592
Source DB: PubMed Journal: Blood ISSN: 0006-4971 Impact factor: 22.113