Successful treatment of B cell chronic lymphocytic leukemia-associated severe paraneoplastic pemphigus with cyclosporin A

Acta Haematol. 2003;109(4):202-5. doi: 10.1159/000070972.

Abstract

Since the first description of paraneoplastic pemphigus, several cases have been described in the literature. However, curative therapy is usually a challenge to the physicians treating this disease. Several publications are available discussing the efficacy of steroids, cyclophosphamide and cyclosporin A. Recently, a report of the successful use of rituximab was also published. However, the use of cyclosporin A is controversial in the case of B cell malignancies, as there are reports showing the cytotoxic effect of this drug on B cells. However, other authors report no effect, or even unwanted effects resulting in B cell proliferation. We report the case of a 50-year-old Caucasian male. He developed a B cell lymphoma consisting of CD5/CD20-double-positive cells, and 2 months later, it was followed by a very severe paraneoplastic pemphigus affecting the mucosa and the skin. The lymphoma was well managed with CHOP and CVP polychemotherapy, followed by oral chlorambucil; however, the bullous eruptions did not disappear. Oral steroids, cyclophosphamide, plasmapheresis and IVIG therapy were only partially successful, so we decided to use oral cyclosporin A. Starting with 7 mg/kg and maintaining a steady plasma level of no less then 110 ng/l, the bullae completely disappeared within 6 weeks, and the patient has been in remission for 17 months now, taking the oral cyclosporin A continuously. The underlying B cell disorder did not relapse during the therapy.

Publication types

  • Case Reports

MeSH terms

  • Antineoplastic Combined Chemotherapy Protocols / administration & dosage
  • Antineoplastic Combined Chemotherapy Protocols / therapeutic use
  • Chlorambucil / administration & dosage
  • Combined Modality Therapy
  • Cyclophosphamide / administration & dosage
  • Cyclosporine / therapeutic use*
  • Doxorubicin / administration & dosage
  • Drug Therapy, Combination
  • Humans
  • Immunosuppressive Agents / therapeutic use*
  • Leukemia, Lymphocytic, Chronic, B-Cell / complications*
  • Leukemia, Lymphocytic, Chronic, B-Cell / drug therapy
  • Male
  • Methylprednisolone / therapeutic use
  • Middle Aged
  • Paraneoplastic Syndromes / drug therapy*
  • Paraneoplastic Syndromes / etiology
  • Paraneoplastic Syndromes / therapy
  • Pemphigus / drug therapy*
  • Pemphigus / etiology
  • Pemphigus / therapy
  • Plasma Exchange
  • Prednisone / administration & dosage
  • Remission Induction
  • Vincristine / administration & dosage

Substances

  • Immunosuppressive Agents
  • Chlorambucil
  • Vincristine
  • Doxorubicin
  • Cyclosporine
  • Cyclophosphamide
  • Prednisone
  • Methylprednisolone

Supplementary concepts

  • CHOP protocol
  • COP protocol 2