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      A Case of Reactive Plasmacytosis Mimicking Multiple Myeloma in A Patient with Primary Sjögren's Syndrome

      case-report

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          Abstract

          Primary Sjögren's syndrome (pSS) is a chronic autoimmune disease with well-documented association of lymphoid malignancies during the progress of the disease. Although several types of malignancy and pseudomalignancy have been reported in pSS, low-grade non-Hodgkin's lymphomas are the most frequently observed. Reactive plasmacytosis mimicking myeloma is a very rare condition in association with pSS. We describe a 72-yr-old woman with pSS who presented with hypergammaglobulinemia, and extensive bone marrow and lymph node plasmacytosis, which mimicked multiple myeloma. In this patient, there was an abnormal differentiation of memory B cells to plasma cells in the peripheral blood suggesting underlying pathogenetic mechanism for this condition.

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          Abnormal B cell differentiation in primary Sjögren's syndrome results in a depressed percentage of circulating memory B cells and elevated levels of soluble CD27 that correlate with Serum IgG concentration.

          M B Bjørgan, Roland Jonsson, Paige K. Thompson … (2002)
          The percentage of CD27(+) B cells in peripheral blood (PB) of patients with primary Sjögren's syndrome (pSS) is significantly decreased compared to normals. In contrast, serum levels of the soluble form of CD27 (sCD27) are significantly higher in pSS patients, with a strong positive correlation between sCD27 and serum IgG levels. In vitro experiments demonstrate that normal B cells cultured under conditions driving plasma cell differentiation result in the production of substantial amounts of sCD27. Analyses of V(H)-region genes from sorted CD27(+) and CD27(-) B cells from pSS patients confirm that the CD27(+) population corresponds to the somatically mutated memory compartment, as in healthy individuals. Together our data indicate that in pSS, there is an abnormal differentiation of B cells to plasma cells resulting in a depression of the circulating memory B-cell pool and the release of significant amounts of sCD27 and IgG.
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            THE DEVELOPMENT OF MALIGNANT LYMPHOMA IN THE COURSE OF SJOEGREN'S SYNDROME.

            J BUNIM, N Talal (1964)
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              Significantly depressed percentage of CD27+ (memory) B cells among peripheral blood B cells in patients with primary Sjögren's syndrome.

              J Thoen, Paige K. Thompson, J Bohnhorst … (2001)
              CD27 has been found to be expressed on somatically mutated B cells and is thus a positive marker for memory B cells in peripheral blood (PB). Since abnormal immunogloblin (Ig) production is characteristic of the autoimmune diseases primary Sjögren's syndrome (pSS) and rheumatoid arthritis (RA), we have analyzed in detail the CD27 expression on PB B cell from these patient groups. Staining of PB B cells with monoclonal antibodies (MoAb) specific for CD19 and CD27 revealed a significantly depressed percentage of CD27+ PB B cells in patients with pSS (14.8 +/- 1.6%) compared to both healthy donors (31.3 +/- 4.7%, P = 0.005) and patients with RA (40.8 +/- 4.1%, P = 0.0001). In addition, the percentages of both the IgD+CD27+ and the IgD-CD27+ B-cell subpopulations were significantly lower in pSS patients compared to RA patients and healthy donors. However, the relative proportion of IgD- and IgD+ cells among the CD27+B cells were almost the same for the three groups. Our data suggest a disturbance in the differentiation of peripheral B cells and possibly a bias towards plasma cell differentiation, resulting in a depressed percentage of CD27+ memory PB B cells in pSS. These results are potentially of pathological significance and of diagnostic value.
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                Author and article information

                Journal
                Journal ID (nlm-ta): J Korean Med Sci
                Journal ID (publisher-id): JKMS
                Title: Journal of Korean Medical Science
                Publisher: The Korean Academy of Medical Sciences
                ISSN (Print): 1011-8934
                ISSN (Electronic): 1598-6357
                Publication date (Print): June 2005
                Publication date (Electronic): 30 June 2005
                Volume: 20
                Issue: 3
                Pages: 506-508
                Affiliations
                Department of Internal Medicine, Ewha Womans University College of Medicine, Seoul, Korea.
                [* ]Department of Pathology, Ewha Womans University College of Medicine, Seoul, Korea.
                Author notes
                Address for correspondence: Jisoo Lee, M.D. Department of Internal Medicine, Ewha Womans University College of Medicine, 911-1 Mok-dong, Yangcheon-gu, Seoul 158-710, Korea. Tel: +82.2-2650-6164, Fax: +82.2-2655-2076, leejisoo@ 123456ewha.ac.kr
                Article
                DOI: 10.3346/jkms.2005.20.3.506
                PMC ID: 2782213
                PubMed ID: 15953879
                SO-VID: 539c0b54-67d2-436a-8114-a155e4cf55bd
                Copyright © Copyright © 2005 The Korean Academy of Medical Sciences
                License:

                This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( http://creativecommons.org/licenses/by-nc/3.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                Date received : 19 March 2004
                Date accepted : 07 June 2004
                Categories
                Subject: Case Report

                ScienceOpen disciplines: Medicine
                Keywords: plasmacytosis,b cell differentiation,sjogren's syndrome,plasma cells
                Data availability:
                ScienceOpen disciplines: Medicine
                Keywords: plasmacytosis, b cell differentiation, sjogren's syndrome, plasma cells

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