title: 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.
authors.affiliation: School of Dentistry and Oral Health, Griffith University, Gold Coast Campus, QLD, Australia; Molecular Basis of Disease Research Program, Griffith Health Institute, Griffith University, Gold Coast Campus, QLD, Australia; Population & Social Health Research Program (Population Oral Health) Griffith Health Institute, Griffith University, Gold Coast Campus, QLD, Australia.
authors.forename: D J
authors.initials: DJ
authors.lastname: Speicher
authors.forename: M M
authors.initials: MM
authors.lastname: Sehu
authors.forename: P
authors.initials: P
authors.lastname: Mollee
authors.forename: L
authors.initials: L
authors.lastname: Shen
authors.forename: N W
authors.initials: NW
authors.lastname: Johnson
authors.forename: J L
authors.initials: JL
authors.lastname: Faoagali
mesh_terms: D058846:Antibodies, Monoclonal, Murine-Derived / Q000627:therapeutic use*; D005871:Castleman Disease / Q000188:drug therapy* / Q000821:virology; D018023:HIV Seronegativity; D006566:Herpesviridae Infections / Q000188:drug therapy* / Q000821:virology; D019288:Herpesvirus 8, Human / Q000302:isolation & purification*; D006801:Humans; D007049:Iatrogenic Disease / Q000517:prevention & control*; D007155:Immunologic Factors / Q000627:therapeutic use; D008107:Liver Diseases / Q000150:complications / Q000601:surgery / Q000821:virology*; D016031:Liver Transplantation; D008297:Male; D008875:Middle Aged; D010802:Phylogeny; D011379:Prognosis; D000069283:Rituximab
chemical_list: D058846:Antibodies, Monoclonal, Murine-Derived; D007155:Immunologic Factors; D000069283:Rituximab
keywords: Castleman's disease; human herpesvirus-8; immunosuppression; liver transplantation; qPCR; rituximab