Thymic plasma cell neoplasm infiltration in Sjögren’s syndrome with monoclonal gammopathy

Thymic plasma cell neoplasm infiltration in Sjögren’s syndrome with monoclonal gammopathy Tommaso Schioppo et al. 23 Cutolo M, Pizzorni C, Tuccio M et al. Nailfold videoca- 25 Airo P, Rossi M, Scarsi M et al. Disease-modifying effects pillaroscopic patterns and serum autoantibodies in of long-term cyclic iloprost therapy in systemic sclerosis. systemic sclerosis. Rheumatology (Oxford) A retrospective analysis and comparison with a control 2004;43:71926. group. Clin Exp Rheumatol 2007;25:7227. 24 Etehad Tavakol M, Fatemi A, Karbalaie A, Emrani Z, 26 Auriemma M, Vianale G, Reale M et al.Iloprosttreat- Erlandsson BE. Nailfold capillaroscopy in rheumatic dis- ment summer-suspension: effects on skin thermal eases: which parameters should be evaluated? Biomed properties and cytokine profile in systemic sclerosis Res Int 2015;2015:974530. patients. G Ital Dermatol Venereol 2013;148:20916. Rheumatology 2018;57:1416 doi:10.1093/rheumatology/key074 Clinical Vignette Advance Access publication 24 March 2018 Thymic plasma cell neoplasm infiltration in FIG.1 CT and MRI and histopathology of mediastinal Sjogren’s syndrome with monoclonal gammopathy mass in SS A 69-year-old woman with SS, which was diagnosed 1 year before, presented with 3-year history of arthralgia and dry mouth, and an anterior mediastinal mass on CT (Fig. 1A, arrow). She had no extraglandular features besides arthritis. The biochemical profile showed elevated levels of CRP (2.2 mg/dl), IgA (2307 mg/dl), anti-SSA/Ro (> 240 U/ml) and anti-SSB/La antibody (20.2 U/ml). Soluble IL-2 receptor was negative. Serum immunoelectrophoresis showed IgA- type M-protein. Bone marrow examination revealed a normal pro- portion of plasma cells (4.1%), indicating monoclonal gam- mopathy of undetermined significance (MGUS). A biopsy from high-intensity anterior mediastinal mass on MRI (fat- suppressed T2) (Fig. 1B, arrow) revealed plasmacytoid cell infiltration of the thymus (Fig. 1C). Immunohistochemistry re- vealed that the infiltrating cells were positive for CD138 (Fig. 1D), IgA and Ig light chain  (Fig. 1E), but negative for light chain . The patient was diagnosed with thymic plasma cell neoplasm infiltration in SS with MGUS. Chemotherapy was initiated, and she was discharged without complica- tions. Plasma cell neoplasm infiltration of the thymus is rare [1]. It has been noted, however, that the presence of MGUS is associated with an increased risk of multiple mye- loma in primary SS [2]. To our knowledge, this is the first Correspondence to: Yoshinori Taniguchi, Department of reported case of plasma cell neoplasm infiltration of the Endocrinology, Metabolism and Nephrology, Kochi Medical thymus in SS with MGUS. This case should remind readers School, Kochi University, Kohasu, Oko-cho, Nankoku, Kochi to consider plasma cell neoplasm infiltration as a rare cause 783-8505, Japan. of mediastinal mass found in SS with MGUS. E-mail: taniguchiy@kochi-u.ac.jp Funding: No specific funding was received from any bodies in the public, commercial or not-for-profit sectors to carry out the work described in this manuscript. References Disclosure statement: The authors have declared no conflicts of interest. 1 Xu YH, Sun LG, Sun C et al. Anterior mediastinum invasion by multiple myeloma: a case report. Oncol Lett 1 1 Yoshinori Taniguchi , Satoshi Inotani and 2017;13:263741. Yoshio Terada 2 Tomi AL, Belkhir R, Nocturne G et al. Monoclonal gam- mopathy and risk of lymphoma and multiple myeloma in Department of Endocrinology, Metabolism, Nephrology and patients with primary Sjo ¨ gren’s syndrome. Arthritis Rheumatology, Kochi Medical School Hospital, Kochi University, Nankoku, Japan Rheumatol 2016;68:124550. ! The Author(s) 2018. Published by Oxford University Press on behalf of the British Society for Rheumatology. All rights reserved. For permissions, please email: journals.permissions@oup.com 1416 https://academic.oup.com/rheumatology Downloaded from https://academic.oup.com/rheumatology/article-abstract/57/8/1416/4953525 by Ed 'DeepDyve' Gillespie user on 24 July 2018 http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Rheumatology Oxford University Press

Thymic plasma cell neoplasm infiltration in Sjögren’s syndrome with monoclonal gammopathy

Rheumatology , Volume Advance Article (8) – Mar 24, 2018
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Oxford University Press
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© The Author(s) 2018. Published by Oxford University Press on behalf of the British Society for Rheumatology. All rights reserved. For permissions, please email: journals.permissions@oup.com
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1462-0324
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1462-0332
D.O.I.
10.1093/rheumatology/key074
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Abstract

Tommaso Schioppo et al. 23 Cutolo M, Pizzorni C, Tuccio M et al. Nailfold videoca- 25 Airo P, Rossi M, Scarsi M et al. Disease-modifying effects pillaroscopic patterns and serum autoantibodies in of long-term cyclic iloprost therapy in systemic sclerosis. systemic sclerosis. Rheumatology (Oxford) A retrospective analysis and comparison with a control 2004;43:71926. group. Clin Exp Rheumatol 2007;25:7227. 24 Etehad Tavakol M, Fatemi A, Karbalaie A, Emrani Z, 26 Auriemma M, Vianale G, Reale M et al.Iloprosttreat- Erlandsson BE. Nailfold capillaroscopy in rheumatic dis- ment summer-suspension: effects on skin thermal eases: which parameters should be evaluated? Biomed properties and cytokine profile in systemic sclerosis Res Int 2015;2015:974530. patients. G Ital Dermatol Venereol 2013;148:20916. Rheumatology 2018;57:1416 doi:10.1093/rheumatology/key074 Clinical Vignette Advance Access publication 24 March 2018 Thymic plasma cell neoplasm infiltration in FIG.1 CT and MRI and histopathology of mediastinal Sjogren’s syndrome with monoclonal gammopathy mass in SS A 69-year-old woman with SS, which was diagnosed 1 year before, presented with 3-year history of arthralgia and dry mouth, and an anterior mediastinal mass on CT (Fig. 1A, arrow). She had no extraglandular features besides arthritis. The biochemical profile showed elevated levels of CRP (2.2 mg/dl), IgA (2307 mg/dl), anti-SSA/Ro (> 240 U/ml) and anti-SSB/La antibody (20.2 U/ml). Soluble IL-2 receptor was negative. Serum immunoelectrophoresis showed IgA- type M-protein. Bone marrow examination revealed a normal pro- portion of plasma cells (4.1%), indicating monoclonal gam- mopathy of undetermined significance (MGUS). A biopsy from high-intensity anterior mediastinal mass on MRI (fat- suppressed T2) (Fig. 1B, arrow) revealed plasmacytoid cell infiltration of the thymus (Fig. 1C). Immunohistochemistry re- vealed that the infiltrating cells were positive for CD138 (Fig. 1D), IgA and Ig light chain  (Fig. 1E), but negative for light chain . The patient was diagnosed with thymic plasma cell neoplasm infiltration in SS with MGUS. Chemotherapy was initiated, and she was discharged without complica- tions. Plasma cell neoplasm infiltration of the thymus is rare [1]. It has been noted, however, that the presence of MGUS is associated with an increased risk of multiple mye- loma in primary SS [2]. To our knowledge, this is the first Correspondence to: Yoshinori Taniguchi, Department of reported case of plasma cell neoplasm infiltration of the Endocrinology, Metabolism and Nephrology, Kochi Medical thymus in SS with MGUS. This case should remind readers School, Kochi University, Kohasu, Oko-cho, Nankoku, Kochi to consider plasma cell neoplasm infiltration as a rare cause 783-8505, Japan. of mediastinal mass found in SS with MGUS. E-mail: taniguchiy@kochi-u.ac.jp Funding: No specific funding was received from any bodies in the public, commercial or not-for-profit sectors to carry out the work described in this manuscript. References Disclosure statement: The authors have declared no conflicts of interest. 1 Xu YH, Sun LG, Sun C et al. Anterior mediastinum invasion by multiple myeloma: a case report. Oncol Lett 1 1 Yoshinori Taniguchi , Satoshi Inotani and 2017;13:263741. Yoshio Terada 2 Tomi AL, Belkhir R, Nocturne G et al. Monoclonal gam- mopathy and risk of lymphoma and multiple myeloma in Department of Endocrinology, Metabolism, Nephrology and patients with primary Sjo ¨ gren’s syndrome. Arthritis Rheumatology, Kochi Medical School Hospital, Kochi University, Nankoku, Japan Rheumatol 2016;68:124550. ! The Author(s) 2018. Published by Oxford University Press on behalf of the British Society for Rheumatology. All rights reserved. For permissions, please email: journals.permissions@oup.com 1416 https://academic.oup.com/rheumatology Downloaded from https://academic.oup.com/rheumatology/article-abstract/57/8/1416/4953525 by Ed 'DeepDyve' Gillespie user on 24 July 2018

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RheumatologyOxford University Press

Published: Mar 24, 2018

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