Juvenile xanthogranuloma with hematological dysfunction treated with 2CDA-AraC

Pediatr Blood Cancer. 2010 Oct;55(4):757-60. doi: 10.1002/pbc.22629.

Abstract

Juvenile xanthogranuloma was diagnosed in two infants aged 8 and 2 months with skin lesions, hepatosplenomegaly, and pancytopenia. Disease control was not achieved with first-line vinblastine-steroid-VP16 combination therapy. Two courses of 2-chlorodeoxyadenosine (2CDA) (0.3 mg/kg) and cytosine arabinoside (AraC) (1 g/m(2)) were then administered for 5 days and were followed, after hematological recovery, by maintenance therapy. Both patients had normal complete blood cell counts and no signs of JXG, respectively, 31 and 24 months after diagnosis.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't
  • Review

MeSH terms

  • Cladribine / administration & dosage*
  • Cytarabine / administration & dosage*
  • Drug Therapy, Combination
  • Female
  • Hematologic Diseases / etiology*
  • Humans
  • Infant
  • Male
  • Xanthogranuloma, Juvenile / blood
  • Xanthogranuloma, Juvenile / drug therapy*

Substances

  • Cytarabine
  • Cladribine