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Severe persistent bone marrow failure following therapy with 2-chlorodeoxyadenosine for relapsing juvenile xanthogranuloma of the brain.

Abstract
2-Chlorodeoxyadenosine (2-CdA) has been successfully used in children to treat refractory Langerhans cell histiocytosis and juvenile xanthogranuloma (JXG) as salvage therapy. Although 2-CdA is generally well-tolerated, with temporary myelosuppression as the primary dose-limiting toxicity, prolonged myelosuppressive, and immunosuppressive effects have been reported. We describe an adolescent patient with refractory multiple central nervous system JXG, with the lesion size markedly reduced after treatment with 2-CdA. However, severe transfusion-dependent bone marrow failure developed after five courses of 2-CdA. He underwent successful bone marrow transplantation from his HLA compatible sister with reduced intensity conditioning.
AuthorsKayo Yamada, Masahiro Yasui, Akihisa Sawada, Masami Inoue, Masahiro Nakayama, Keisei Kawa
JournalPediatric blood & cancer (Pediatr Blood Cancer) Vol. 58 Issue 2 Pg. 300-2 (Feb 2012) ISSN: 1545-5017 [Electronic] United States
PMID21384540 (Publication Type: Journal Article)
CopyrightCopyright © 2011 Wiley Periodicals, Inc.
Chemical References
  • Immunosuppressive Agents
  • Cladribine
Topics
  • Anemia, Aplastic
  • Bone Marrow Diseases
  • Bone Marrow Failure Disorders
  • Bone Marrow Transplantation
  • Brain Diseases (complications, therapy)
  • Child
  • Cladribine (adverse effects)
  • Hemoglobinuria, Paroxysmal (etiology, therapy)
  • Humans
  • Immunosuppressive Agents (adverse effects)
  • Male
  • Prognosis
  • Recurrence
  • Transfusion Reaction
  • Xanthogranuloma, Juvenile (complications, therapy)

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