Journal
PEDIATRIC BLOOD & CANCER
Volume 57, Issue 7, Pages 1239-1243Publisher
WILEY PERIODICALS, INC
DOI: 10.1002/pbc.23226
Keywords
hemophagocytic lymphohistiocytosis; LCMV; rapamycin; sirolimus
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- XLP Research Trust
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Hemophagocytic lymphohistiocytosis (HLH) is an immunodysregulatory disorder for which more effective treatments are needed. The macrolide rapamycin has immunosuppressive properties, making it an attractive candidate for controlling the aberrant T cell activation that occurs in HLH. To investigate its therapeutic potential, we used rapamycin to treat Lymphocytic Choriomeningitis Virus (LCMV)-infected perforin-deficient (Prf1(-/-)) mice according to a well-established model of HLH. At the regimens tested, rapamycin did not improve weight loss, splenomegaly, hemophagocytosis, cytopenias, or proinflammatory cytokine production in LCMV-infected Prf1(-/-) animals. Thus, single agent rapamycin appears ineffective in treating the clinical and laboratory manifestations of LCMV-induced HLH. Pediatr Blood Cancer 2011;57:1239-1243. (C) 2011 Wiley Periodicals, Inc.
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