Literature DB >> 15284122

Mutations of the Wiskott-Aldrich Syndrome Protein (WASP): hotspots, effect on transcription, and translation and phenotype/genotype correlation.

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.

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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


  101 in total

1.  Ubiquitylation-dependent negative regulation of WASp is essential for actin cytoskeleton dynamics.

Authors:  Barak Reicher; Noah Joseph; Ahuvit David; Maor H Pauker; Orly Perl; Mira Barda-Saad
Journal:  Mol Cell Biol       Date:  2012-06-04       Impact factor: 4.272

2.  Developmental expression of Drosophila Wiskott-Aldrich Syndrome family proteins.

Authors:  Evelyn Rodriguez-Mesa; Maria Teresa Abreu-Blanco; Alicia E Rosales-Nieves; Susan M Parkhurst
Journal:  Dev Dyn       Date:  2012-01-31       Impact factor: 3.780

Review 3.  Wiskott-Aldrich syndrome: another piece in the puzzle.

Authors:  L D Notarangelo; L Mori
Journal:  Clin Exp Immunol       Date:  2005-02       Impact factor: 4.330

4.  Impaired cell adhesion, apoptosis, and signaling in WASP gene-disrupted Nalm-6 pre-B cells and recovery of cell adhesion using a transducible form of WASp.

Authors:  Rikiya Sato; Susumu Iiizumi; Eun-Sung Kim; Fumiko Honda; Sang-Kyou Lee; Noritaka Adachi; Hideki Koyama; Shuki Mizutani; Tomohiro Morio
Journal:  Int J Hematol       Date:  2012-02-05       Impact factor: 2.490

5.  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

6.  Transcellular diapedesis is initiated by invasive podosomes.

Authors:  Christopher V Carman; Peter T Sage; Tracey E Sciuto; Miguel A de la Fuente; Raif S Geha; Hans D Ochs; Harold F Dvorak; Ann M Dvorak; Timothy A Springer
Journal:  Immunity       Date:  2007-06       Impact factor: 31.745

Review 7.  Warts and all: human papillomavirus in primary immunodeficiencies.

Authors:  Jennifer W Leiding; Steven M Holland
Journal:  J Allergy Clin Immunol       Date:  2012-10-01       Impact factor: 10.793

8.  Clinical characteristics and genetic profiles of 44 patients with severe combined immunodeficiency (SCID): report from Shanghai, China (2004-2011).

Authors:  Chun-Mei Yao; Xiao-Hua Han; Yi-Dan Zhang; Hui Zhang; Ying-Ying Jin; Rui-Ming Cao; Xi Wang; Quan-Hua Liu; Wei Zhao; Tong-Xin Chen
Journal:  J Clin Immunol       Date:  2012-12-19       Impact factor: 8.317

Review 9.  Inherited platelet disorders: thrombocytopenias and thrombocytopathies.

Authors:  Giovanna D'Andrea; Massimiliano Chetta; Maurizio Margaglione
Journal:  Blood Transfus       Date:  2009-10       Impact factor: 3.443

10.  Rapid platelet turnover in WASP(-) mice correlates with increased ex vivo phagocytosis of opsonized WASP(-) platelets.

Authors:  Amanda Prislovsky; Bindumadhav Marathe; Amira Hosni; Alyssa L Bolen; Falk Nimmerjahn; Carl W Jackson; Darryl Weiman; Ted S Strom
Journal:  Exp Hematol       Date:  2008-03-17       Impact factor: 3.084

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