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Successful treatment of iatrogenic multicentric Castleman's disease arising due to recrudescence of HHV-8 in a liver transplant patient.

Abstract
We describe the case of a 59-year-old HIV-negative male who developed multicentric Castleman's disease (MCD) 1 year postliver transplantation due to recrudescence of a pretransplant human herpesvirus-8 (HHV-8) infection. He presented with fevers, dry cough, weight loss and drenching night sweats. Routine investigations were all unremarkable. Computerized axial tomography (CT) scans showed splenomegaly and intra-abdominal lymphadenopathy, confirmed by positron emission tomography. Cervical lymph node biopsies were consistent with MCD. The presence of HHV-8 was confirmed on immunohistochemistry. Peripheral blood HHV-8 quantitative polymerase chain reaction (qPCR) monitoring showed a threefold decrease in viremia in the first week of treatment with ganciclovir but had little impact on clinical symptoms. Reducing immunosuppression and switching to rituximab resolved clinical symptoms and produced a negative HHV-8 qPCR result. Retrospective molecular testing of sera collected pre- and immediately posttransplantation confirmed preexisting HHV-8 in the host. This is the first reported case of an HIV-negative postliver transplant patient developing MCD that manifested as posttransplant lymphoproliferative disorder due to recrudescence of HHV-8. We propose (1) the introduction of the term iatrogenic Castleman's disease (CD) for this and similar cases, (2) rituximab should be considered as a treatment option for CD and (3) consideration be given to a change to the World Health Organization classification of CD to incorporate such cases.
AuthorsD J Speicher, M M Sehu, P Mollee, L Shen, N W Johnson, J L Faoagali
JournalAmerican journal of transplantation : official journal of the American Society of Transplantation and the American Society of Transplant Surgeons (Am J Transplant) Vol. 14 Issue 5 Pg. 1207-13 (May 2014) ISSN: 1600-6143 [Electronic] United States
PMID24674650 (Publication Type: Case Reports, Journal Article)
Copyright© Copyright 2014 The American Society of Transplantation and the American Society of Transplant Surgeons.
Chemical References
  • Antibodies, Monoclonal, Murine-Derived
  • Immunologic Factors
  • Rituximab
Topics
  • Antibodies, Monoclonal, Murine-Derived (therapeutic use)
  • Castleman Disease (drug therapy, virology)
  • HIV Seronegativity
  • Herpesviridae Infections (drug therapy, virology)
  • Herpesvirus 8, Human (isolation & purification)
  • Humans
  • Iatrogenic Disease (prevention & control)
  • Immunologic Factors (therapeutic use)
  • Liver Diseases (complications, surgery, virology)
  • Liver Transplantation
  • Male
  • Middle Aged
  • Phylogeny
  • Prognosis
  • Rituximab

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