Epstein-Barr virus-positive B-cell lymphoproliferative disorders arising in immunodeficient patients previously treated with fludarabine for low-grade B-cell neoplasms

Am J Surg Pathol. 2002 May;26(5):630-6. doi: 10.1097/00000478-200205000-00009.

Abstract

We describe five patients with treated low-grade B-cell neoplasms who subsequently developed Epstein-Barr virus (EBV)-positive B-cell lymphoproliferative disorders (BLPDs). The low-grade B-cell neoplasms were B-cell chronic lymphocytic leukemia in four patients and splenic marginal zone lymphoma in one patient. All patients had received treatment with fludarabine for the low-grade B-cell neoplasm, and three had also received Campath-1H. The EBV-BLPDs arose 2-12 months after completion of fludarabine therapy and morphologically resembled the EBV-BLPDs that occur in the setting of iatrogenic immunodeficiency. Molecular genetic studies showed that these lesions were clonally distinct from the low-grade B-cell neoplasm in three of four cases assessed. Two patients did not receive therapy for the EBV-BLPD. The lesions regressed spontaneously in both patients but recurred in one. One patient underwent surgical excision and remains without evidence of the EBV-BLPD. One patient received aggressive multiagent chemotherapy with a complete response initially, but the EBV-BLPD recurred after 12 months. One patient received antiviral therapy and responded completely but died 2 months later of an opportunistic infection. We conclude that patients with low-grade B-cell neoplasms treated with fludarabine, possibly in combination with other immune suppressive agents, may subsequently develop EBV-BLPDs that morphologically resemble other iatrogenic immunodeficiency-associated BLPDs. Most are clonally distinct from the underlying low-grade B-cell neoplasm. A subset of these lesions may regress without systemic therapy.

MeSH terms

  • Antineoplastic Agents / adverse effects*
  • Epstein-Barr Virus Infections / chemically induced*
  • Epstein-Barr Virus Infections / pathology
  • Epstein-Barr Virus Infections / virology
  • Female
  • Herpesvirus 4, Human / genetics
  • Herpesvirus 4, Human / isolation & purification*
  • Humans
  • Immunocompromised Host*
  • Immunosuppressive Agents / adverse effects*
  • Lymphoma, B-Cell / drug therapy*
  • Lymphoma, Non-Hodgkin / drug therapy*
  • Lymphoproliferative Disorders / chemically induced*
  • Lymphoproliferative Disorders / pathology
  • Lymphoproliferative Disorders / virology
  • Male
  • RNA-Binding Proteins / analysis
  • Ribosomal Proteins*
  • Vidarabine / adverse effects*
  • Vidarabine / analogs & derivatives

Substances

  • Antineoplastic Agents
  • Immunosuppressive Agents
  • RNA-Binding Proteins
  • Ribosomal Proteins
  • RPL22 protein, human
  • Vidarabine
  • fludarabine