title: A case of nodular sclerosis Hodgkin's lymphoma repeatedly relapsing in the context of composite plasma cell-hyaline vascular Castleman's disease: successful response to rituximab and radiotherapy.
abstract: We report the case of an Epstein-Barr virus (EBV)- and human immunodeficiency virus-serum negative patient suffering from repeatedly relapsing classical Hodgkin's Lymphoma (cHL) associated with a histological picture of plasma cell-hyaline vascular (PC-HV) form of Castleman's disease (CD). The CD30- and CD15-positive, Reed-Sternberg/Hodgkin cells, only occasionally expressed the CD20 molecule, but not leukocyte common antigen and latent membrane protein-1. Single-strand polymerase chain reaction failed to detect human herpesvirus 8 or EBV in the involved tissues. At the time of second relapse in July 2005, the clinical picture was characterized by a palpable right hypogastric mass, disclosed at physical exam, in the absence of other enlarged peripheral lymph nodes, subjective symptoms or laboratory profile alterations. Combined hybrid-(18)F-fluorodeoxyglucose positron emission-computerized tomography (18F-FDG PET/CT) showed increased radionuclide uptake in multiple external iliac lymph nodes [standardized uptake value (SUV) of 7.4] and non-palpable left supraclavicular lymph nodes (SUV of 5.8). Relapsing cHL in the context of mixed PC-HV CD was documented in two of three surgically excised abdominal lymph nodes never previously enlarged or involved by any lymphoproliferative disease. Because of the limited disease extension and failure to induce continuous remission with previous conventional chemoradiotherapy, the patient was treated with six rituximab injections. This immunotherapy induced significant reduction in size of supraclavicular lymph nodes as evident at ultrasound (US) scan (<1 vs. 2.5 cm, post- vs. pretherapy), which was confirmed by the 18F-FDG PET/CT in October 2005, despite no modification in SUV of 4.2. 18F-FDG PET/CT also disclosed no radionuclide uptake by abdominal lymph nodes. Thus, a second course of four additional rituximab injections was given and subsequent 18F-FDG PET/CT indicated persistent, but reduced incorporation of radionuclide compared to the pretherapy value (SUV of 2.7) in the supraclavicular area and confirmed a normal metabolic activity in the iliac external lymph nodes. Because of uncertain persistent disease in the supraclavicular nodal site, involved-field radiotherapy (RT) was delivered in that area as consolidation treatment. After completion of rituximab and RT for 16 and 14 months respectively, US and 18F-FDG PET/CT exams were indicative of complete remission. This case is in concordance with previously published data suggesting that rituximab immunotherapy might be a valid option in the treatment of CD and also have a role in the management of relapsing cHL.
authors: Section of Internal Medicine and Oncologic Sciences, Policlinico Monteluce, Perugia, Italy.
authors: Lorenzo
authors: L
authors: Falchi
authors: Daniela
authors: D
authors: Capello
authors: Barbara
authors: B
authors: Palumbo
authors: Annamaria
authors: A
authors: Rauco
authors: Rita
authors: R
authors: Emili
authors: Michele
authors: M
authors: Cianciulli
authors: Roberta
authors: R
authors: Pace
authors: Vincenzo
authors: V
authors: Capparella
authors: Fabrizio
authors: F
authors: Liberati
authors: Anna Marina
authors: AM
authors: Liberati
mesh_terms: D000328:Adult; D000911:Antibodies, Monoclonal / Q000627:therapeutic use*; D058846:Antibodies, Monoclonal, Murine-Derived; D000970:Antineoplastic Agents / Q000627:therapeutic use*; D005871:Castleman Disease / Q000150:complications*; D017024:Chemotherapy, Adjuvant; D005260:Female; D019788:Fluorodeoxyglucose F18; D006689:Hodgkin Disease / Q000150:complications / Q000175:diagnosis / Q000188:drug therapy* / Q000378:metabolism / Q000473:pathology / Q000532:radiotherapy*; D006801:Humans; D006818:Hyalin / Q000378:metabolism*; D007155:Immunologic Factors / Q000627:therapeutic use*; D010950:Plasma Cells; D049268:Positron-Emission Tomography; D019275:Radiopharmaceuticals; D018714:Radiotherapy, Adjuvant; D012008:Recurrence; D000069283:Rituximab; D014057:Tomography, X-Ray Computed; D016896:Treatment Outcome
chemical_list: D000911:Antibodies, Monoclonal; D058846:Antibodies, Monoclonal, Murine-Derived; D000970:Antineoplastic Agents; D007155:Immunologic Factors; D019275:Radiopharmaceuticals; D019788:Fluorodeoxyglucose F18; D000069283:Rituximab