共 13 条
Use of Sirolimus (Rapamycin) for Treatment of Cytopenias and Lymphoproliferation Linked to Autoimmune Lymphoproliferative Syndrome (ALPS). Two Case Reports
被引:7
作者:

Cayrol, Julie
论文数: 0 引用数: 0
h-index: 0
机构:
Gregorio Maranon Childrens Hosp, Dept Pediat Hematol Oncol, Biomed Res Inst, Madrid, Spain
Royal Childrens Hosp, Childrens Canc Ctr, Melbourne, Vic, Australia Gregorio Maranon Childrens Hosp, Dept Pediat Hematol Oncol, Biomed Res Inst, Madrid, Spain

Colino, Carmen Garrido
论文数: 0 引用数: 0
h-index: 0
机构:
Gregorio Maranon Childrens Hosp, Dept Pediat Hematol Oncol, Biomed Res Inst, Madrid, Spain Gregorio Maranon Childrens Hosp, Dept Pediat Hematol Oncol, Biomed Res Inst, Madrid, Spain
机构:
[1] Gregorio Maranon Childrens Hosp, Dept Pediat Hematol Oncol, Biomed Res Inst, Madrid, Spain
[2] Royal Childrens Hosp, Childrens Canc Ctr, Melbourne, Vic, Australia
关键词:
autoimmune lymphoproliferative syndrome;
lymphoproliferation;
cytopenias;
sirolimus;
SYNDROME ALPS;
D O I:
10.1097/MPH.0000000000000785
中图分类号:
R73 [肿瘤学];
学科分类号:
100214 ;
摘要:
Autoimmune lymphoproliferative syndrome (ALPS) is a disorder of lymphocyte apoptosis. Children present with chronic nonmalignant lymphadenopathy, hepatosplenomegaly, and autoimmune cytopenias. Recent advances show efficacy of treatment with immunosuppressive drugs. Sirolimus, an mammalian target of rapamycin inhibitor, improves autoimmune cytopenias and lymphoproliferation, with a safe profile. We present 2 patients, a 5-year-old girl and 15-year-old boy, diagnosed with ALPS with initial partial response to steroid treatment. Autoimmune cytopenias and lymphoproliferation then became refractory to treatment, with recurrence of symptoms. In both cases, treatment with sirolimus was started, with a rapid response, complete remission of cytopenias, and resolution of lymphoproliferation, with no significant adverse effects. Conclusion: sirolimus is an effective and safe drug for controlling children with cytopenias and lymphoproliferation linked to ALPS.
引用
收藏
页码:E187 / E190
页数:4
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h-index: 0
机构:
NIAID, Immunol Lab, NIH, Bethesda, MD 20892 USA NIAID, Immunol Lab, NIH, Bethesda, MD 20892 USA

Chun, HJ
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NIAID, Immunol Lab, NIH, Bethesda, MD 20892 USA NIAID, Immunol Lab, NIH, Bethesda, MD 20892 USA

Lenardo, MJ
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NIAID, Immunol Lab, NIH, Bethesda, MD 20892 USA NIAID, Immunol Lab, NIH, Bethesda, MD 20892 USA