Sirolimus
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Cytomegalovirus flare: case report A male patient [exact age at the time of reaction onset not stated] developed flare of cytomegalovirus infection during treatment with unspecified steroids and sirolimus. The male patient was presented with autoimmune haemolytic anaemia, splenomegaly, axillary adenopathy and viral pneumonia at the age of 3 years. He had developed massive hepatosplenomegaly with refractory thrombocytopenia despite treatment with unspecified immunosuppressive therapy including steroids along with various medications. Then, he developed cytomegalovirus infection. Three years later, he developed acquired amegakaryocytic thrombocytopenia and acquired pure red cell aplasia. His acquired amegakaryocytic thrombocytopenia and acquired pure red cell aplasia were responsive to treatment with unspecified immunosuppressive therapy including highdose steroids and sirolimus. However, he developed cytomegalovirus infection flare secondary to unspecified immunosuppressive therapy including high-dose steroids and sirolimus [dosage, route and duration of treatment to reaction onset not stated]. Subsequent investigation led to the diagnosis of lipopolysaccharide-responsive beige-like anchor (LRBA) protein deficiency due to a homozygous splice site mutation, c.6364-1G >C [outcome not stated]. Author comment: "His [acquired pure red cell aplasia] and [acquired amegakaryocytic thrombocytopenia] were responsive to increases in [immunosuppressive therapy] (highdose steroids with addition of sirolimus); however, this led to a flare of his [cytomegalovirus] disease." Rajpurkar M, et al. Acquired Pure Red Cell Aplasia and Acquired Amegakaryocytic Thrombocytopenia Associated with Clonal Expansion of T-Cell Large Granular Lymphocytes in a Patient with Lipopolysaccharide-responsive Beige-like Anchor (LRBA) Protein Deficiency. Journal of Pediatric Hematology/ 803439093 Oncology 41: E542-E545, No. 8, Nov 2019 - USA
0114-9954/19/1783-0001/$14.95 Adis © 2019 Springer Nature Switzerland AG. All rights reserved
Reactions 14 Dec 2019 No. 1783
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