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Nishikori, Asami Department of Molecular Hematopathology, Okayama University Graduate School of Health Sciences
Nishimura, Midori Filiz Department of Molecular Hematopathology, Okayama University Graduate School of Health Sciences
Nishimura, Yoshito Department of General Medicine, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences ORCID publons researchmap
Otsuka, Fumio Department of General Medicine, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences ORCID Kaken ID publons researchmap
Maehama, Kanna Department of Molecular Hematopathology, Okayama University Graduate School of Health Sciences
Ohsawa, Kumiko Department of Pathology, Saitama Medical Center, Saitama Medical University
Momose, Shuji Department of Pathology, Saitama Medical Center, Saitama Medical University
Nakamura, Naoya Department of Pathology, Tokai University School of Medicine
Sato, Yasuharu Department of Molecular Hematopathology, Okayama University Graduate School of Health Sciences ORCID Kaken ID researchmap
Abstract
Idiopathic multicentric Castleman disease (iMCD) is a type of Castleman disease that is not related to KSHV/HHV8 infection. Currently, iMCD is classified into iMCD-TAFRO (thrombocytopenia, anasarca, fever, reticulin fibrosis, and organomegaly) and iMCD-NOS (not otherwise specified). The former has been established as a relatively homogeneous disease unit that has been recently re-defined, while the latter is considered to be a heterogeneous disease that could be further divided into several subtypes. In 1980, Mori et al. proposed the concept of idiopathic plasmacytic lymphadenopathy (IPL), a disease presenting with polyclonal hypergammaglobulinemia and a sheet-like proliferation of mature plasma cells in the lymph nodes. Some researchers consider IPL to be a part of iMCD-NOS, although it has not been clearly defined to date. This is the first paper to analyze iMCD-NOS clinicopathologically, to examine whether IPL forms a uniform disease unit in iMCD. Histologically, the IPL group showed prominent plasmacytosis and the hyperplasia of germinal centers, while the non-IPL group showed prominent vascularity. Clinically, the IPL group showed significant thrombocytosis and elevated serum IgG levels compared to the non-IPL group (p = 0.007, p < 0.001, respectively). Pleural effusion and ascites were less common in the IPL group (p < 0.001). The IPL group was more likely to have an indolent clinical course and a good response to the anti-IL-6 receptor antibody, while the non-IPL counterpart frequently required more aggressive medical interventions. Thus, the IPL group is a clinicopathologically uniform entity that forms an independent subtype of iMCD.
Keywords
Castleman disease
idiopathic multicentric Castleman disease
idiopathic plasmacytic lymphadenopathy
plasma cell morphology
Published Date
2022-09-07
Publication Title
International Journal Of Molecular Sciences
Volume
volume23
Issue
issue18
Publisher
MDPI
Start Page
10301
ISSN
1422-0067
Content Type
Journal Article
language
English
OAI-PMH Set
岡山大学
Copyright Holders
© 2022 by the authors.
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PubMed ID
DOI
Web of Science KeyUT
Related Url
isVersionOf https://doi.org/10.3390/ijms231810301
License
https://creativecommons.org/licenses/by/4.0/
Funder Name
Japan Society for the Promotion of Science
助成番号
JP 20K07407