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Prepublished online as a Blood First Edition Paper on July 5, 2002; DOI 10.1182/blood-2002-02-0487.
Submitted February 15, 2002
Accepted April 15, 2002
KSHV and EBV associated germinotropic lymphoproliferative disorder
Ming-Qing Du*, Tim C Diss, Hongxiang Liu, Hongtao Ye, Rifat A Hamoudi, Jose Cabecadas, Henry Y Dong, Nancy Lee Harris, John K C Chan, John W Rees, Ahmet Dogan, and Peter G Isaacson
Department of Histopathology, University College London, London, United Kingdom
Servico de Anatomia Patologica, Instituto Portugues de Oncologia de Francisco Gentil, Lisbon, Portugal
Department of Pathology, Massachusetts General Hospital, Boston, USA
Department of Pathology, Queen Elizabeth Hospital, Hong Kong, China
Department of Pathology, Dandenong Hospital, Victoria, Australia
* Corresponding author; email: m.du{at}ucl.ac.uk.
Kaposi's sarcoma-associated herpesvirus (KSHV) is known to be associated with three distinct lymphoproliferative disorders: primary effusion lymphoma (PEL), multicentric Castleman's disease (MCD) and MCD-associated plasmablastic lymphoma. We report three cases of a previously undescribed KSHV associated lymphoproliferative disorder. The disease presented as localized lymphadenopathy and showed a favorable response to chemo- or radiotherapy. Histologically, the lymphoproliferation is characterized by plasmablasts that preferentially involved germinal centers of the lymphoid follicles, forming confluent aggregates. They were negative for CD20, CD27, CD79a, CD138, BCL6 and CD10, but showed monotypic  or  light chain. Clusters of CD10+ CD20+ residual follicle center cells were identified in some of the follicles. The plasmablasts were positive for both KSHV and EBV, and most of them also expressed viral IL6. Unexpectedly, molecular analysis of whole tissue sections or microdissected KSHV positive aggregates demonstrated a poly- or oligoclonal pattern of Ig gene rearrangement. The plasmablasts showed somatic mutation and intraclonal variation in the rearranged Ig genes, and one case expressed switched Ig heavy chain (IgA), suggesting that they originated from germinal center B-cells. We propose calling this distinctive entity "KSHV associated germinotropic lymphoproliferative disorder."
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