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.
Am J Transplant. 2014 May;14(5):1207-13. doi: 10.1111/ajt.12693. Epub 2014 Mar 26.
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.
我们描述了一例 59 岁的 HIV 阴性男性患者，在肝移植后 1 年因移植前人类疱疹病毒 8 型（HHV-8）感染复发而发生多中心 Castleman 病（MCD）。他表现为发热、干咳、体重减轻和盗汗。常规检查均无异常。计算机轴向断层扫描（CT）显示脾肿大和腹腔内淋巴结病，正电子发射断层扫描（PET）进一步证实。颈部淋巴结活检符合 MCD。免疫组化证实存在 HHV-8。外周血 HHV-8 定量聚合酶链反应（qPCR）监测显示，在使用更昔洛韦治疗的第一周，病毒血症下降了三倍，但对临床症状影响不大。减少免疫抑制并改用利妥昔单抗可缓解临床症状，并使 HHV-8 qPCR 结果转为阴性。对移植前和移植后立即采集的血清进行回顾性分子检测证实了宿主中存在预先存在的 HHV-8。这是首例报道的 HIV 阴性肝移植后患者发生 MCD 的病例，表现为因 HHV-8 复发而引起的移植后淋巴组织增生性疾病。我们提出（1）将这种情况和类似情况命名为医源性 Castleman 病（CD），（2）利妥昔单抗应被视为 CD 的治疗选择，以及（3）考虑将 CD 的世界卫生组织分类进行修改，纳入此类病例。
