Acral Skin Rash Caused by Altered Mercaptopurine Metabolism in Maintenance Therapy for B-Cell Acute Lymphoblastic Leukemia

J Pediatr Hematol Oncol. 2022 Oct 1;44(7):398-401. doi: 10.1097/MPH.0000000000002434. Epub 2022 Feb 18.

Abstract

6-mercaptopurine is a mainstay of acute lymphoblastic leukemia treatment. It has a narrow therapeutic window, dictated by its metabolite, thioguanine and 6-methylmercaptopurine. Skin manifestations usually consist of mild facial rash or hypersensitivity exanthems. We report a child who developed a painful acral rash and mucositis while undergoing maintenance therapy for B-cell acute lymphoblastic leukemia without infectious or known drug etiology. Thiopurine metabolites were skewed toward 6-methylmercaptopurine. Two weeks after allopurinol was added and 6-mercaptopurine (6-MP) dose adjusted, the cutaneous manifestations and other constitutional symptoms resolved. We posit that the rash was because of 6-MP toxicity related to skewed metabolism, adding to the growing list of toxicity related to altered 6-MP metabolism.

MeSH terms

  • Allopurinol / therapeutic use
  • Burkitt Lymphoma* / drug therapy
  • Child
  • Exanthema* / chemically induced
  • Humans
  • Mercaptopurine / analogs & derivatives
  • Precursor Cell Lymphoblastic Leukemia-Lymphoma* / drug therapy
  • Precursor Cell Lymphoblastic Leukemia-Lymphoma* / metabolism
  • Thioguanine / metabolism
  • Thioguanine / therapeutic use

Substances

  • Allopurinol
  • 6-methylthiopurine
  • Mercaptopurine
  • Thioguanine