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Prepublished online as a Blood First Edition Paper on July 5, 2002; DOI 10.1182/blood-2002-02-0487.
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Blood, 1 November 2002, Vol. 100, No. 9, pp. 3415-3418
BRIEF REPORT
KSHV- and EBV-associated germinotropic lymphoproliferative
disorder
Ming-Qing Du,
Tim C. Diss,
Hongxiang Liu,
Hongtao Ye,
Rifat A. Hamoudi,
José Cabeçadas,
Henry Y. Dong,
Nancy Lee Harris,
John K. C. Chan,
John W. Rees,
Ahmet Dogan, and
Peter G. Isaacson
From the Department of Histopathology, University
College London, London, United Kingdom; Serviço de Anatomia
Patológica, Instituto Português de Oncologia de Francisco
Gentil, Lisbon, Portugal; Department of Pathology,
Massachusetts General Hospital, Boston, MA; Department of Pathology,
Queen Elizabeth Hospital, Hong Kong; and Department of Pathology,
Dandenong Hospital, Victoria, Australia.
Kaposi sarcoma-associated herpesvirus (KSHV) is known to be
associated with 3 distinct lymphoproliferative disorders: primary effusion lymphoma (PEL), multicentric Castleman disease (MCD), and
MCD-associated plasmablastic lymphoma. We report 3 cases of a
previously undescribed KSHV-associated lymphoproliferative disorder. The disease presented as localized lymphadenopathy and showed a
favorable response to chemotherapy 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
interleukin-6 (vIL-6). Unexpectedly, molecular analysis of whole tissue
sections or microdissected KSHV-positive aggregates demonstrated a
polyclonal or oligoclonal pattern of immunoglobulin (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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