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Research ArticleArticle

Bidirectional Relationship Between Primary Sjögren Syndrome and Non-Hodgkin Lymphoma: A Nationwide Taiwanese Population-based Study

Li-Hui Wang, Wei-Ming Wang, Chun-Yu Lin, Sheng-Hsiang Lin and Chi-Chang Shieh
The Journal of Rheumatology January 2020, jrheum.191027; DOI: https://doi.org/10.3899/jrheum.191027
Li-Hui Wang
From the Division of Immunology, Department of Pediatrics, Kuo General Hospital; Institute of Clinical Medicine, College of Medicine, National Cheng Kung University; Division of Allergy, Immunology and Rheumatology, and Department of Pediatrics, Department of Internal Medicine, and Department of Statistics, National Cheng Kung University Hospital, Tainan, Taiwan. Supported by a grant from Kuo General Hospital. L.H. Wang, MD, MS, Immunologist, Division of Immunology, Department of Pediatrics, Kuo General Hospital; W.M. Wang, MS, Biostatistician, National Cheng Kung University, Department of Statistics; C.Y. Lin, MD, Rheumatologist, National Cheng Kung University Hospital, Department of Internal Medicine; S.H. Lin, PhD, Associate Professor of Biostatistics, National Cheng Kung University College of Medicine, Institute of Clinical Medicine; C.C. Shieh, MD, PhD, Professor of Immunology, Rheumatology, and Pediatrics, National Cheng Kung University College of Medicine, Institute of Clinical Medicine. Address correspondence to Dr. C.C. Shieh, Institute of Clinical Medicine, National Cheng-Kung University, 138 Sheng-Li Road, Tainan 70403, Taiwan. E-mail: cshieh@mail.ncku.edu.tw. Accepted for publication January 7, 2020.
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Wei-Ming Wang
From the Division of Immunology, Department of Pediatrics, Kuo General Hospital; Institute of Clinical Medicine, College of Medicine, National Cheng Kung University; Division of Allergy, Immunology and Rheumatology, and Department of Pediatrics, Department of Internal Medicine, and Department of Statistics, National Cheng Kung University Hospital, Tainan, Taiwan. Supported by a grant from Kuo General Hospital. L.H. Wang, MD, MS, Immunologist, Division of Immunology, Department of Pediatrics, Kuo General Hospital; W.M. Wang, MS, Biostatistician, National Cheng Kung University, Department of Statistics; C.Y. Lin, MD, Rheumatologist, National Cheng Kung University Hospital, Department of Internal Medicine; S.H. Lin, PhD, Associate Professor of Biostatistics, National Cheng Kung University College of Medicine, Institute of Clinical Medicine; C.C. Shieh, MD, PhD, Professor of Immunology, Rheumatology, and Pediatrics, National Cheng Kung University College of Medicine, Institute of Clinical Medicine. Address correspondence to Dr. C.C. Shieh, Institute of Clinical Medicine, National Cheng-Kung University, 138 Sheng-Li Road, Tainan 70403, Taiwan. E-mail: cshieh@mail.ncku.edu.tw. Accepted for publication January 7, 2020.
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Chun-Yu Lin
From the Division of Immunology, Department of Pediatrics, Kuo General Hospital; Institute of Clinical Medicine, College of Medicine, National Cheng Kung University; Division of Allergy, Immunology and Rheumatology, and Department of Pediatrics, Department of Internal Medicine, and Department of Statistics, National Cheng Kung University Hospital, Tainan, Taiwan. Supported by a grant from Kuo General Hospital. L.H. Wang, MD, MS, Immunologist, Division of Immunology, Department of Pediatrics, Kuo General Hospital; W.M. Wang, MS, Biostatistician, National Cheng Kung University, Department of Statistics; C.Y. Lin, MD, Rheumatologist, National Cheng Kung University Hospital, Department of Internal Medicine; S.H. Lin, PhD, Associate Professor of Biostatistics, National Cheng Kung University College of Medicine, Institute of Clinical Medicine; C.C. Shieh, MD, PhD, Professor of Immunology, Rheumatology, and Pediatrics, National Cheng Kung University College of Medicine, Institute of Clinical Medicine. Address correspondence to Dr. C.C. Shieh, Institute of Clinical Medicine, National Cheng-Kung University, 138 Sheng-Li Road, Tainan 70403, Taiwan. E-mail: cshieh@mail.ncku.edu.tw. Accepted for publication January 7, 2020.
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Sheng-Hsiang Lin
From the Division of Immunology, Department of Pediatrics, Kuo General Hospital; Institute of Clinical Medicine, College of Medicine, National Cheng Kung University; Division of Allergy, Immunology and Rheumatology, and Department of Pediatrics, Department of Internal Medicine, and Department of Statistics, National Cheng Kung University Hospital, Tainan, Taiwan. Supported by a grant from Kuo General Hospital. L.H. Wang, MD, MS, Immunologist, Division of Immunology, Department of Pediatrics, Kuo General Hospital; W.M. Wang, MS, Biostatistician, National Cheng Kung University, Department of Statistics; C.Y. Lin, MD, Rheumatologist, National Cheng Kung University Hospital, Department of Internal Medicine; S.H. Lin, PhD, Associate Professor of Biostatistics, National Cheng Kung University College of Medicine, Institute of Clinical Medicine; C.C. Shieh, MD, PhD, Professor of Immunology, Rheumatology, and Pediatrics, National Cheng Kung University College of Medicine, Institute of Clinical Medicine. Address correspondence to Dr. C.C. Shieh, Institute of Clinical Medicine, National Cheng-Kung University, 138 Sheng-Li Road, Tainan 70403, Taiwan. E-mail: cshieh@mail.ncku.edu.tw. Accepted for publication January 7, 2020.
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Chi-Chang Shieh
From the Division of Immunology, Department of Pediatrics, Kuo General Hospital; Institute of Clinical Medicine, College of Medicine, National Cheng Kung University; Division of Allergy, Immunology and Rheumatology, and Department of Pediatrics, Department of Internal Medicine, and Department of Statistics, National Cheng Kung University Hospital, Tainan, Taiwan. Supported by a grant from Kuo General Hospital. L.H. Wang, MD, MS, Immunologist, Division of Immunology, Department of Pediatrics, Kuo General Hospital; W.M. Wang, MS, Biostatistician, National Cheng Kung University, Department of Statistics; C.Y. Lin, MD, Rheumatologist, National Cheng Kung University Hospital, Department of Internal Medicine; S.H. Lin, PhD, Associate Professor of Biostatistics, National Cheng Kung University College of Medicine, Institute of Clinical Medicine; C.C. Shieh, MD, PhD, Professor of Immunology, Rheumatology, and Pediatrics, National Cheng Kung University College of Medicine, Institute of Clinical Medicine. Address correspondence to Dr. C.C. Shieh, Institute of Clinical Medicine, National Cheng-Kung University, 138 Sheng-Li Road, Tainan 70403, Taiwan. E-mail: cshieh@mail.ncku.edu.tw. Accepted for publication January 7, 2020.
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Abstract

Objective Bidirectional relationships between some autoimmune diseases and non-Hodgkin lymphoma (NHL) may exist. We conducted this nationwide population-based study in Taiwan to investigate whether there is a bidirectional relationship between primary Sjögren syndrome (pSS) and NHL.

Methods Using the National Health Insurance Research Database of Taiwan, we identified 15,636 patients with new-onset pSS without previous cancer and 25,074 patients with new-onset NHL without previous pSS as 2 non-overlapping cohorts from 1998 to 2012, and followed them until 2013. Standardized incidence ratios (SIR) for NHL in the patients with pSS and SIR for pSS in the patients with NHL were compared with the general population.

Results Among the 15,636 patients with pSS, 741 developed cancers, including 51 with NHL. The highest SIR of specific cancer risk in patients with pSS was that for NHL (SIR 4.6, 95% CI 3.4–6.0). Among the 25,074 patients with NHL, 49 developed pSS; the SIR was also increased (SIR 3.2, 95% CI 2.4–4.2). The risk was highest within 1 year after the diagnosis of each disease.

Conclusion This nationwide population-based study is the first to report a bidirectional relationship between pSS and NHL. Our findings suggest being alert for patients with pSS or NHL who have early signs of the other disease in clinical care. The underlying mechanisms of the bidirectional relationship merit further investigation.

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Bidirectional Relationship Between Primary Sjögren Syndrome and Non-Hodgkin Lymphoma: A Nationwide Taiwanese Population-based Study
Li-Hui Wang, Wei-Ming Wang, Chun-Yu Lin, Sheng-Hsiang Lin, Chi-Chang Shieh
The Journal of Rheumatology Jan 2020, jrheum.191027; DOI: 10.3899/jrheum.191027

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Bidirectional Relationship Between Primary Sjögren Syndrome and Non-Hodgkin Lymphoma: A Nationwide Taiwanese Population-based Study
Li-Hui Wang, Wei-Ming Wang, Chun-Yu Lin, Sheng-Hsiang Lin, Chi-Chang Shieh
The Journal of Rheumatology Jan 2020, jrheum.191027; DOI: 10.3899/jrheum.191027
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