Literature DB >> 9697838

Wiskott-Aldrich syndrome protein-deficient mice reveal a role for WASP in T but not B cell activation.

S B Snapper1, F S Rosen, E Mizoguchi, P Cohen, W Khan, C H Liu, T L Hagemann, S P Kwan, R Ferrini, L Davidson, A K Bhan, F W Alt.   

Abstract

The Wiskott-Aldrich syndrome (WAS) is a human X-linked immunodeficiency resulting from mutations in a gene (WASP) encoding a cytoplasmic protein implicated in regulating the actin cytoskeleton. To elucidate WASP function, we disrupted the WASP gene in mice by gene-targeted mutation. WASP-deficient mice showed apparently normal lymphocyte development, normal serum immunoglobulin levels, and the capacity to respond to both T-dependent and T-independent type II antigens. However, these mice did have decreased peripheral blood lymphocyte and platelet numbers and developed chronic colitis. Moreover, purified WASP-deficient T cells showed markedly impaired proliferation and antigen receptor cap formation in response to anti-CD3epsilon stimulation. Yet, purified WASP-deficient B cells showed normal responses to anti-Ig stimulation. We discuss the implications of our findings regarding WASP function in receptor signaling and cytoskeletal reorganization in T and B cells and compare the effects of WASP deficiency in mice and humans.

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Year:  1998        PMID: 9697838     DOI: 10.1016/s1074-7613(00)80590-7

Source DB:  PubMed          Journal:  Immunity        ISSN: 1074-7613            Impact factor:   31.745


  158 in total

Review 1.  The Wiskott-Aldrich syndrome.

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

2.  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

3.  Systemic autoimmunity and defective Fas ligand secretion in the absence of the Wiskott-Aldrich syndrome protein.

Authors:  Nikolay P Nikolov; Masaki Shimizu; Sophia Cleland; Daniel Bailey; Joseph Aoki; Ted Strom; Pamela L Schwartzberg; Fabio Candotti; Richard M Siegel
Journal:  Blood       Date:  2010-05-10       Impact factor: 22.113

4.  Defective inhibition of B-cell proliferation by Wiskott-Aldrich syndrome protein-deficient regulatory T cells.

Authors:  Marsilio Adriani; Krysten A Jones; Toru Uchiyama; Martha R Kirby; Christopher Silvin; Stacie M Anderson; Fabio Candotti
Journal:  Blood       Date:  2011-04-22       Impact factor: 22.113

5.  Actin cytoskeleton regulates calcium dynamics and NFAT nuclear duration.

Authors:  Fabiola V Rivas; James P O'Keefe; Maria-Luisa Alegre; Thomas F Gajewski
Journal:  Mol Cell Biol       Date:  2004-02       Impact factor: 4.272

Review 6.  Mechanisms of intestinal inflammation and development of associated cancers: lessons learned from mouse models.

Authors:  Aya M Westbrook; Akos Szakmary; Robert H Schiestl
Journal:  Mutat Res       Date:  2010-03-16       Impact factor: 2.433

7.  B cell-intrinsic deficiency of the Wiskott-Aldrich syndrome protein (WASp) causes severe abnormalities of the peripheral B-cell compartment in mice.

Authors:  Mike Recher; Siobhan O Burns; Miguel A de la Fuente; Stefano Volpi; Carin Dahlberg; Jolan E Walter; Kristin Moffitt; Divij Mathew; Nadine Honke; Philipp A Lang; Laura Patrizi; Hervé Falet; Marton Keszei; Masayuki Mizui; Eva Csizmadia; Fabio Candotti; Kari Nadeau; Gerben Bouma; Ottavia M Delmonte; Francesco Frugoni; Angela B Ferraz Fomin; David Buchbinder; Emma Maria Lundequist; Michel J Massaad; George C Tsokos; John Hartwig; John Manis; Cox Terhorst; Raif S Geha; Scott Snapper; Karl S Lang; Richard Malley; Lisa Westerberg; Adrian J Thrasher; Luigi D Notarangelo
Journal:  Blood       Date:  2012-02-01       Impact factor: 22.113

8.  Lymphocyte-dependent and Th2 cytokine-associated colitis in mice deficient in Wiskott-Aldrich syndrome protein.

Authors:  Deanna D Nguyen; Michel H Maillard; Vinicius Cotta-de-Almeida; Emiko Mizoguchi; Christoph Klein; Ivan Fuss; Cathryn Nagler; Atsushi Mizoguchi; Atul K Bhan; Scott B Snapper
Journal:  Gastroenterology       Date:  2007-07-12       Impact factor: 22.682

Review 9.  Rheumatologic and autoimmune manifestations of primary immunodeficiency disorders.

Authors:  Ramona Goyal; Ariel C Bulua; Nikolay P Nikolov; Pamela L Schwartzberg; Richard M Siegel
Journal:  Curr Opin Rheumatol       Date:  2009-01       Impact factor: 5.006

Review 10.  Genetically engineered mouse models for studying inflammatory bowel disease.

Authors:  Atsushi Mizoguchi; Takahito Takeuchi; Hidetomo Himuro; Toshiyuki Okada; Emiko Mizoguchi
Journal:  J Pathol       Date:  2015-11-14       Impact factor: 7.996

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