| Literature DB >> 9697838 |
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.Entities:
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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