Abstract
The extent, range, and nature of available research in the field of primary Sjögren’s syndrome (pSS) have not been understood fully. This study aimed to map the literature available on pSS, and identify global hotspots and trends in the research. Papers on pSS published between 2004 and 2021 were searched from Web of Science Core Collection. The quantity and citations of publications, and the research hotspots and trends in the field of pSS were analyzed and presented visually by Microsoft Excel and Citespace software. A total of 3606 papers mainly from 526 institutions in 83 countries/regions were included for analysis. The number of publications presented an overall upward trend in the field of pSS from 2004 to 2021. The USA ranked first in the number of publications (n = 661), followed by China (n = 491), Italy (n = 405), France (n = 351), and Japan (n = 292). Moreover, seven of the top ten countries by the number of publications on pSS were from Europe. The University of Groningen (n = 661), Xavier Mariette (n = 95), and Clinical and Experimental Rheumatology (n = 184) were the most prolific affiliation, author, and journal, respectively. Vitali C (n = 2009) and Arthritis and Rheumatism (n = 3918) held the record for the most cited papers by an author and journal, respectively. At present, the hot keywords in the field of pSS include disease activity, ultrasonography, management, consensus, and data-driven. Lymphoid organization, clinical phenotypes outcome, salivary gland ultrasonography, and Toll-like receptor are the emerging research trends in pSS. Research on pSS is flourishing. Current research of pSS mainly focuses on disease activity, ultrasonography, and management. While, the emerging research trends in pSS are lymphoid organization, clinical phenotypes outcome, salivary gland ultrasonography, and Toll-like receptor.
Similar content being viewed by others
Data availability
Data sharing is not applicable to this article as no new data were created in this study. These datasets were derived from the following public domain resources: https://www.webofscience.com/wos/woscc/advanced-search.
References
Bloch KJ, Buchanan WW, Wohl MJ, Bunim JJ (1965) Sjöegren’s Syndrome. a clinical, pathological, and serological study of sixty-two cases. Medicine (Baltimore) 44:187–231
Nocturne G, Mariette X (2018) B cells in the pathogenesis of primary Sjögren syndrome. Nat Rev Rheumatol 14:133–145. https://doi.org/10.1038/nrrheum.2018.1
Mariette X, Criswell LA (2018) Primary Sjögren’s Syndrome. N Engl J Med 378:931–939. https://doi.org/10.1056/NEJMcp1702514
Patel R, Shahane A (2014) The epidemiology of Sjögren’s syndrome. Clin Epidemiol 6:247–255. https://doi.org/10.2147/CLEP.S47399
Zhang X, Feng R, Zhao J, Wang Y, He J, Liu L, Cheng Y, Yao H, Tang S, Chen J, Zhang S, Zhang Z, Wang Q, He J, Li Z (2021) Salivary gland ultrasonography in primary Sjögren’s syndrome from diagnosis to clinical stratification: a multicentre study. Arthritis Res Ther 23:305. https://doi.org/10.1186/s13075-021-02689-3
Vasaitis L, Nordmark G, Theander E, Backlin C, Smedby KE, Askling J, Rönnblom L, Sundström C, Baecklund E (2020) Population-based study of patients with primary Sjögren’s syndrome and lymphoma: lymphoma subtypes, clinical characteristics, and gender differences. Scand J Rheumatol 49:225–232. https://doi.org/10.1080/03009742.2019.1696403
Brito-Zerón P, Retamozo S, Kostov B, Baldini C, Bootsma H, De Vita S, Dörner T, Gottenberg JE, Kruize AA, Mandl T, Ng WF, Seror R, Tzioufas AG, Vitali C, Bowman S, Mariette X, Ramos-Casals M (2019) Efficacy and safety of topical and systemic medications: a systematic literature review informing the EULAR recommendations for the management of Sjögren’s syndrome. RMD Open 5:e001064. https://doi.org/10.1136/rmdopen-2019-001064
Tzioufas AG, Kapsogeorgou EK, Moutsopoulos HM (2012) Pathogenesis of Sjögren’s syndrome: what we know and what we should learn. J Autoimmun 39:4–8. https://doi.org/10.1016/j.jaut.2012.01.002
Fox RI, Fox CM, Gottenberg JE, Dörner T (2021) Treatment of Sjögren’s syndrome: current therapy and future directions. Rheumatology (Oxford) 60:2066–2074. https://doi.org/10.1093/rheumatology/kez142
Seror R, Nocturne G, Mariette X (2021) Current and future therapies for primary Sjögren syndrome. Nat Rev Rheumatol 17:475–486. https://doi.org/10.1038/s41584-021-00634-x
Chi Y (2013) Global trends in medical journal publishing. J Korean Med Sci 28:1120–1121. https://doi.org/10.3346/jkms.2013.28.8.1120
Han Y, Wennersten SA, Lam MPY (2019) Working the literature harder: what can text mining and bibliometric analysis reveal? Expert Rev Proteomics 16:871–873. https://doi.org/10.1080/14789450.2019.1703678
Chen C (2006) CiteSpace II: Detecting and visualizing emerging trends and transient patterns in scientific literature. J Americ Soc Inform Sci Technol 57:359–377. https://doi.org/10.1002/asi.20317
Akyol A, Kocyigit BF (2021) Publication activity in the field of Sjögren’s syndrome: a ten-year web of science based analysis. Rheumatol Int 41:763–769. https://doi.org/10.1007/s00296-020-04714-1
Chen C (2004) Searching for intellectual turning points: progressive knowledge domain visualization. Proc Natl Acad Sci U S A 101:5303–5310. https://doi.org/10.1073/pnas.0307513100
Akyol A, Kocyigit BF (2021) Ankylosing spondylitis rehabilitation publications and the global productivity: a web of science-based bibliometric analysis (2000–2019). Rheumatol Int 41:2007–2014. https://doi.org/10.1007/s00296-021-04836-0
Kocyigit BF, Akyol A (2021) Bibliometric and altmetric analyses of publication activity in the field of Behcet’s disease in 2010–2019. J Korean Med Sci 36:e207. https://doi.org/10.3346/jkms.2021.36.e207
Bournia VK, Vlachoyiannopoulos PG (2012) Subgroups of Sjögren syndrome patients according to serological profiles. J Autoimmun 39:15–26
Brito-Zerón P, Baldini C, Bootsma H, Bowman SJ, Jonsson R, Mariette X, Sivils K, Theander E, Tzioufas A, Ramos-Casals M (2016) Sjögren syndrome. Nat Rev Dis Primers 2:16047. https://doi.org/10.1038/nrdp.2016.47
Seror R, Ravaud P, Mariette X, Bootsma H, Theander E, Hansen A, Ramos-Casals M, Dörner T, Bombardieri S, Hachulla E, Brun JG, Kruize AA, Praprotnik S, Tomsic M, Gottenberg JE, Devauchelle V, Devita S, Vollenweider C, Mandl T, Tzioufas A, Carsons S, Saraux A, Sutcliffe N, Vitali C, Bowman S, Sjögren’s Task Force EULAR (2011) EULAR Sjogren’s syndrome patient reported index (ESSPRI): development of a consensus patient index for primary Sjogren’s syndrome. Ann Rheum Dis 70:968–972. https://doi.org/10.1136/ard.2010.143743
Seror R, Ravaud P, Bowman SJ, Baron G, Tzioufas A, Theander E, Gottenberg JE, Bootsma H, Mariette X, Vitali C, Sjögren’s Task Force EULAR (2010) EULAR Sjogren’s syndrome disease activity index: development of a consensus systemic disease activity index for primary Sjogren’s syndrome. Ann Rheum Dis 69:1103–1109. https://doi.org/10.1136/ard.2009.110619
Cornec D, Jousse-Joulin S, Pers JO, Marhadour T, Cochener B, Boisrame-Gastrin S, Nowak E, Youinou P, Saraux A, Devauchelle-Pensec V (2013) Contribution of salivary gland ultrasonography to the diagnosis of Sjogren’s syndrome: toward new diagnostic criteria? Arthritis Rheum 65:216–225. https://doi.org/10.1002/art.37698
Theander E, Mandl T (2014) Primary Sjogren’s syndrome: diagnostic and prognostic value of salivary gland ultrasonography using a simplified scoring system. Arthritis Care Res 66:1102–1107. https://doi.org/10.1002/acr.22264
Giovelli RA, Santos MC, Serrano ÉV, Valim V (2015) Clinical characteristics and biopsy accuracy in suspected cases of Sjögren’s syndrome referred to labial salivary gland biopsy. BMC Musculoskelet Disord 16:30. https://doi.org/10.1186/s12891-015-0482-9
Astorri E, Sutcliffe N, Richards PS, Suchak K, Pitzalis C, Bombardieri M, Tappuni AR (2016) Ultrasound of the salivary glands is a strong predictor of labial gland biopsy histopathology in patients with sicca symptoms. J Oral Pathol Med 45:450–454. https://doi.org/10.1111/jop.12387
Hildebrand JM, Luo Z, Manske MK, Price-Troska T, Ziesmer SC, Lin W, Hostager BS, Slager SL, Witzig TE, Ansell SM, Cerhan JR, Bishop GA, Novak AJ (2010) A BAFF-R mutation associated with non-Hodgkin lymphoma alters TRAF recruitment and reveals new insights into BAFF-R signaling. J Exp Med 207:2569–2579. https://doi.org/10.1084/jem.20100857
Goulabchand R, Malafaye N, Jacot W, Witkowski Durand Viel P, Morel J, Lukas C, Rozier P, Lamure S, Noel D, Molinari N, Mura T, Guilpain P (2021) Cancer incidence in primary Sjögren’s syndrome: data from the French hospitalization database. Autoimmun Rev 20:102987. https://doi.org/10.1016/j.autrev.2021.102987
Rivière E, Pascaud J, Tchitchek N, Boudaoud S, Paoletti A, Ly B, Dupré A, Chen H, Thai A, Allaire N, Jagla B, Mingueneau M, Nocturne G, Mariette X (2020) Salivary gland epithelial cells from patients with Sjögren’s syndrome induce B-lymphocyte survival and activation. Ann Rheum Dis 79:1468–1477. https://doi.org/10.1136/annrheumdis-2019-216588
Chen W, Yang F, Xu G, Ma J, Lin J (2021) Follicular helper T cells and follicular regulatory T cells in the immunopathology of primary Sjögren’s syndrome. J Leukoc Biol 109:437–447. https://doi.org/10.1002/JLB.5MR1020-057RR
Kiripolsky J, Kramer JM (2018) Current and emerging evidence for toll-like receptor activation in Sjögren’s syndrome. J Immunol Res 2018:1246818. https://doi.org/10.1155/2018/1246818
Davidson JA, Banerjee A, Smeeth L, McDonald HI, Grint D, Herrett E, Forbes H, Pebody R, Warren-Gash C (2021) Risk of acute respiratory infection and acute cardiovascular events following acute respiratory infection among adults with increased cardiovascular risk in England between 2008 and 2018: a retrospective, population-based cohort study. Lancet Digit Health 3:e773–e783. https://doi.org/10.1016/S2589-7500(21)00203-X
Yin AL, Guo WL, Sholle ET, Rajan M, Alshak MN, Choi JJ, Goyal P, Jabri A, Li HA, Pinheiro LC, Wehmeyer GT, Weiner M (2022) Weill Cornell COVID-19 data abstraction consortium, Safford MM, campion TR, Cole CL. Comparing automated vs. manual data collection for COVID-specific medications from electronic health records. Int J Med Inform 157:104622. https://doi.org/10.1016/j.ijmedinf.2021.104622
Hickman SE, Woitek R, Le EPV, Im YR, Mouritsen Luxhøj C, Aviles-Rivero AI, Baxter GC, MacKay JW, Gilbert FJ (2022) Machine learning for workflow applications in screening mammography: systematic review and meta-analysis. Radiology 302:88–104. https://doi.org/10.1148/radiol.2021210391
Acknowledgements
The authors would like to thank Dr. Guohua Yang for language editing of the manuscript, and Prof. Chaomei Chen for answering questions on ScienceNet.cn.
Funding
This work was supported by National Key Clinical Specialty Capacity Building Project (2011-ZDZK-001), Capital’s Funds for Health Improvement and Research (2020-4-40610), and Elite Medical Professionals project of China–Japan Friendship Hospital (ZRJY2021-QM14).
Author information
Authors and Affiliations
Contributions
Conception and design of the work—JL and QWT: developed the search strategy—JQC and QH: data acquisition, analysis, and interpretation—JQC, QH, BHJ, WJS, ZHW, JYY, ZWH, and XBY: Original draft preparation—JQC: critical revision of the draft—JL, QWT, QH, BHJ, WJS, ZHW, JYY, ZWH, and XBY: all authors give approval of the final version to be published and take full responsibility for the integrity and accuracy of all aspects of the work.
Corresponding authors
Ethics declarations
Conflict of interest
The authors declare that they have no conflict of interest.
Additional information
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Supplementary Information
Below is the link to the electronic supplementary material.
Rights and permissions
About this article
Cite this article
Chen, J., He, Q., Jiang, B. et al. Research on primary Sjögren’s syndrome in 2004–2021: a Web of Science-based cross-sectional bibliometric analysis. Rheumatol Int 42, 2221–2229 (2022). https://doi.org/10.1007/s00296-022-05138-9
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00296-022-05138-9