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Sirolimus as maintenance treatment in an infant with life-threatening multiresistant pure red cell anemia/autoimmune hemolytic anemia.

Abstract
A 9-month-old boy with life-threatening multiresistant pure red cell anemia/autoimmune hemolytic anemia within the frame of a possible, undiagnosed immune-mediated disease was initially treated with prednisone. Further-line therapies of the following 7 relapses included immunoglobulins, rituximab, cyclophosphamide, and alentuzumab followed by other maintenance treatments as cyclosporine, methotrexate, and mycophenolate. After all the administered therapies failed, the patient was successfully treated by splenectomy followed by fludarabine and then sirolimus as maintenance treatment. Relapses might have been caused by the lack of a complete debulking of triggering cells and/or ineffective maintenance therapy. Splenectomy and sirolimus may have played a complementary role in the management of both situations.
AuthorsMaurizio Miano, Vincenzo Poggi, Laura Banov, Francesca Fioredda, Concetta Micalizzi, Johanna Svahn, Giovanni Montobbio, Federica Gallicola, Angelo C Molinari, Rosanna Parasole, Fara Petruzziello, Alain Fischer, Michaela Calvillo, Carlo Dufour
JournalJournal of pediatric hematology/oncology (J Pediatr Hematol Oncol) Vol. 36 Issue 3 Pg. e145-8 (Apr 2014) ISSN: 1536-3678 [Electronic] United States
PMID23588343 (Publication Type: Case Reports, Journal Article)
Chemical References
  • Vidarabine
  • fludarabine
  • Sirolimus
Topics
  • Anemia, Hemolytic, Autoimmune (diagnosis, therapy)
  • Antineoplastic Combined Chemotherapy Protocols (therapeutic use)
  • Combined Modality Therapy
  • Drug Resistance
  • Humans
  • Infant
  • Male
  • Red-Cell Aplasia, Pure (diagnosis, therapy)
  • Sirolimus (administration & dosage)
  • Splenectomy
  • Treatment Outcome
  • Vidarabine (administration & dosage, analogs & derivatives)

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