Cyclosporine A for chemotherapy-resistant subcutaneous panniculitis-like T cell lymphoma with hemophagocytic syndrome

Acta Haematol. 2011;126(1):8-12. doi: 10.1159/000323565. Epub 2011 Mar 17.

Abstract

Subcutaneous panniculitis-like T cell lymphoma (SPTL) is a rare subtype of non-Hodgkin lymphoma for which a definitive therapeutic strategy has not been established yet. We report a case of chemotherapy-resistant SPTL with hemophagocytic syndrome (HPS) which was successfully treated with cyclosporine A (CsA) plus methylprednisolone (mPSL), and also reviewed 11 SPTL cases treated with CsA, previously reported in the literature. Our patient was a 38-year-old female with SPTL. The disease progressed despite conventional chemotherapy using cytotoxic agents including alkylators, anthracyclins or purine analogues, and, after 2 months of chemotherapy, was eventually complicated by HPS and disseminated intravascular coagulation (DIC). CsA (4 mg/kg/day) plus mPSL treatment dramatically improved HPS with DIC, reduced subcutaneous tumors within 2 weeks, and finally induced complete remission (CR) after 3 months. Currently, the patient has maintained CR while being treated with CsA for 12 months. In addition to our case, 9 of 11 SPTL cases were successfully treated with CsA, and 8 were induced to CR. Time to first response to CsA was within 2 weeks in most cases, regardless of prior treatment or the co-occurrence of HPS. Our case and this first comprehensive review on CsA for SPTL suggest that CsA may constitute a candidate treatment strategy for SPTL.

Publication types

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

MeSH terms

  • Adult
  • Antineoplastic Agents / therapeutic use
  • Cyclosporine / therapeutic use*
  • Drug Resistance, Neoplasm
  • Female
  • Humans
  • Lymphohistiocytosis, Hemophagocytic / complications*
  • Lymphoma, T-Cell / complications
  • Lymphoma, T-Cell / diagnostic imaging
  • Lymphoma, T-Cell / drug therapy
  • Panniculitis / complications
  • Panniculitis / diagnostic imaging
  • Panniculitis / drug therapy
  • Positron-Emission Tomography

Substances

  • Antineoplastic Agents
  • Cyclosporine

Supplementary concepts

  • Subcutaneous panniculitis-like T-cell lymphoma