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Phenotypic differences between mice deficient in XIAP and SAP, two factors targeted in X-linked lymphoproliferative syndrome (XLP).

Abstract：

:Mutations in the X-linked inhibitor of apoptosis (XIAP) have recently been identified in patients with the rare genetic disease, X-linked lymphoproliferative syndrome (XLP), which was previously thought to be solely attributable to mutations in a distinct gene, SAP. To further understand the roles of these two factors in the pathogenesis of XLP, we have compared mice deficient in Xiap with known phenotypes of Sap-null mice. We show here that in contrast to Sap-deficient mice, animals lacking Xiap have apparently normal NKT cell development and no apparent defect in humoral responses to T cell-dependent antigens. However, Xiap-deficient cells were more susceptible to death upon infection with the murine herpesvirus MHV-68 and gave rise to more infectious virus. These differences could be rescued by restoration of XIAP. These data provide insight into the differing roles of XIAP and SAP in the pathogenesis of XLP.

journal_name

Cell Immunol

journal_title

Cellular immunology

authors

Rumble JM,Oetjen KA,Stein PL,Schwartzberg PL,Moore BB,Duckett CS

doi

10.1016/j.cellimm.2009.05.017

subject

Has Abstract

pub_date

2009-01-01 00:00:00

pages

82-9

issue

1

eissn

0008-8749

issn

1090-2163

pii

S0008-8749(09)00105-1

journal_volume

259

pub_type

杂志文章

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