Xanthoma disseminatum: effective therapy with 2-chlorodeoxyadenosine in a case series

Arch Dermatol. 2011 Apr;147(4):459-64. doi: 10.1001/archdermatol.2010.378. Epub 2010 Dec 20.

Abstract

Background: Xanthoma disseminatum is a rare nonfamilial disease characterized by lipid deposition in skin and internal organs due to histiocytic cell proliferation, classified as a benign non-Langerhans cell histiocytosis. This chronic disease has no known established treatment. We report 8 cases of xanthoma disseminatum with treatment outcomes in long-term follow-up and provide a review of the medical literature.

Observations: We studied clinical manifestations; disease course; histopathologic and immunopathological findings; and responses to treatment in 8 consecutively seen patients with xanthoma disseminatum, including follow-up. The follow-up period ranged from 3 months to 8 years. Five patients received 2-chlorodeoxyadenosine. Therapy with 2-chlorodeoxyadenosine was effective at inducing remission and long-term control of cutaneous lesions of xanthoma disseminatum in 5 cases. Adverse effects were minimal, and treatment was well tolerated. No improvement was noted in untreated patients.

Conclusion: Among 8 cases of xanthoma disseminatum, a positive response to treatment with 2-chlorodeoxyadenosine was seen in 5 cases.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Aged
  • Chronic Disease
  • Cladribine / therapeutic use*
  • Female
  • Histiocytes / drug effects
  • Histiocytes / pathology
  • Histiocytosis, Non-Langerhans-Cell / drug therapy*
  • Histiocytosis, Non-Langerhans-Cell / pathology
  • Humans
  • Immunosuppressive Agents / therapeutic use*
  • Male
  • Middle Aged
  • Treatment Outcome

Substances

  • Immunosuppressive Agents
  • Cladribine