ArticleJournal of hematopathology2026
HIV-negative KSHV/HHV8-associated multicentric Castleman disease with concurrent Kaposi sarcoma and POEMS-like manifestations.
Article in Journal of hematopathology, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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Abstract
introductionCastleman disease (CD) is a heterogeneous lymphoproliferative disorder with unicentric and multicentric forms. Multicentric CD (MCD) is etiologically classified into idiopathic MCD (iMCD), POEMS-associated MCD, and Kaposi sarcoma-associated herpesvirus (KSHV/HHV-8)-associated MCD, and it typically presents with systemic inflammation. KSHV/HHV-8-associated MCD is most commonly seen in HIV-positive patients, although it may rarely occur in HIV-negative individuals. CASE PRESENTATION: We report a case of a 65-year-old HIV-negative woman diagnosed with mixed-type KSHV/HHV-8-associated MCD accompanied by Kaposi sarcoma and POEMS-like clinical features. The patient presented with generalized lymphadenopathy, splenomegaly, cytopenias, hypercalcemia, and monoclonal IgG lambda gammopathy. PET-CT revealed widespread hypermetabolic lymphadenopathy and a sclerotic bone lesion in the left humerus. Bone marrow examination was normocellular and showed polytypic plasma cell proliferation. Lymph node biopsy demonstrated HHV-8 (LANA-1) positivity consistent with mixed-type MCD and Kaposi sarcoma. HHV-8 DNA positivity was also confirmed.
conclusionThe patient was treated with a regimen including daratumumab, bortezomib, cyclophosphamide, and dexamethasone, achieving a clinical and hematologic response after the third cycle. This case highlights that KSHV/HHV-8-associated MCD may occur in HIV-negative individuals and may present concurrently with Kaposi sarcoma and POEMS-like manifestations, representing a rare diagnostic challenge.
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