- Volume 46 Issue 2
DOI QR Code
A Case Report of Primary Sjögren's Syndrome Presenting Oral Mucosal Pain
- Lee, Ji-Young (Department of Oral Medicine, School of Dentistry, Kyungpook National University) ;
- Byun, Jin-Seok (Department of Oral Medicine, School of Dentistry, Kyungpook National University) ;
- Jung, Jae-Kwang (Department of Oral Medicine, School of Dentistry, Kyungpook National University) ;
- Choi, Jae-Kap (Department of Oral Medicine, School of Dentistry, Kyungpook National University)
- Received : 2021.05.11
- Accepted : 2021.06.06
- Published : 2021.06.30
Sjögren's syndrome (SS) is an autoimmune rheumatic disease characterized by the presence of lymphocytic infiltration of exocrine glands and other organs. Due to the rarity of SS, it is frequently overlooked by dentists as a cause of xerostomia. Clinicians should be aware that patients may have oral complications from SS including oral mucosal pain caused by frictional damage. Use of the latest diagnostic criteria approved by the American College of Rheumatology/European League against Rheumatism in 2016 for SS diagnosis provides the advantage of early identification of patients who have not yet developed dry mouth symptoms. A case of primary SS with the chief complaint of oral mucosal pain caused by mucosal damage associated with dry mouth is presented here. This case represents the importance of recognizing oral complications of xerostomia caused by SS, and illustrates use of the latest diagnostic criteria to diagnose the suspected symptoms by dental clinicians, who typically might be the first medical professional to encounter these symptoms.
- Kassan SS, Moutsopoulos HM. Clinical manifestations and early diagnosis of Sjogren syndrome. Arch Intern Med 2004;164:1275-1284. https://doi.org/10.1001/archinte.164.12.1275
- Brandt JE, Priori R, Valesini G, Fairweather D. Sex differences in Sjogren's syndrome: a comprehensive review of immune mechanisms. Biol Sex Differ 2015;6:19. https://doi.org/10.1186/s13293-015-0037-7
- Cartee DL, Maker S, Dalonges D, Manski MC. Sjogren's syndrome: oral manifestations and treatment, a dental perspective. J Dent Hyg 2015;89:365-371.
- Franceschini F, Cavazzana I, Andreoli L, Tincani A. The 2016 classification criteria for primary Sjogren's syndrome: what's new? BMC Med 2017;15:69. https://doi.org/10.1186/s12916-017-0837-1
- Ramos-Casals M, Tzioufas AG, Font J. Primary Sjogren's syndrome: new clinical and therapeutic concepts. Ann Rheum Dis 2005;64:347-354. https://doi.org/10.1136/ard.2004.025676
- Pertovaara M, Korpela M, Uusitalo H, et al. Clinical follow up study of 87 patients with sicca symptoms (dryness of eyes or mouth, or both). Ann Rheum Dis 1999;58:423-427. https://doi.org/10.1136/ard.58.7.423
- Baer AN, Walitt B. Sjogren syndrome and other causes of sicca in older adults. Clin Geriatr Med 2017;33:87-103.
- Zintzaras E, Voulgarelis M, Moutsopoulos HM. The risk of lymphoma development in autoimmune diseases: a meta-analysis. Arch Intern Med 2005;165:2337-2344. https://doi.org/10.1001/archinte.165.20.2337
- Mariette X, Criswell LA. Primary Sjogren's syndrome. N Engl J Med 2018;378:931-939. https://doi.org/10.1056/NEJMcp1702514
- Villa A, Connell CL, Abati S. Diagnosis and management of xerostomia and hyposalivation. Ther Clin Risk Manag 2014;11:45-51. https://doi.org/10.2147/TCRM.S76282
- Ship JA. Diagnosing, managing, and preventing salivary gland disorders. Oral Dis 2002;8:77-89. https://doi.org/10.1034/j.1601-0825.2002.2o837.x
- Cohen-Brown G, Ship JA. Diagnosis and treatment of salivary gland disorders. Quintessence Int 2004;35:108-123.
- Lopez-Pintor RM, Casanas E, Gonzalez-Serrano J, et al. Xerostomia, hyposalivation, and salivary flow in diabetes patients. J Diabetes Res 2016;2016:4372852.
- Shiboski CH, Shiboski SC, Seror R, et al. 2016 American College of Rheumatology/European League Against Rheumatism classification criteria for primary Sjogren's syndrome: a consensus and data-driven methodology involving three international patient cohorts. Ann Rheum Dis 2017;76:9-16. https://doi.org/10.1136/annrheumdis-2016-210571
- Vitali C, Bombardieri S, Jonsson R, et al. Classification criteria for Sjogren's syndrome: a revised version of the European criteria proposed by the American-European Consensus Group. Ann Rheum Dis 2002;61:554-558. https://doi.org/10.1136/ard.61.6.554
- Shiboski SC, Shiboski CH, Criswell L, et al. American College of Rheumatology classification criteria for Sjogren's syndrome: a data-driven, expert consensus approach in the Sjogren's International Collaborative Clinical Alliance cohort. Arthritis Care Res (Hoboken) 2012;64:475-487. https://doi.org/10.1002/acr.21591
- Cornec D, Saraux A, Cochener B, et al. Level of agreement between 2002 American-European Consensus Group and 2012 American College of Rheumatology classification criteria for Sjogren's syndrome and reasons for discrepancies. Arthritis Res Ther 2014;16:R74. https://doi.org/10.1186/ar4514
- Rasmussen A, Ice JA, Li H, et al. Comparison of the AmericanEuropean Consensus Group Sjogren's syndrome classification criteria to newly proposed American College of Rheumatology criteria in a large, carefully characterised sicca cohort. Ann Rheum Dis 2014;73:31-38 https://doi.org/10.1136/annrheumdis-2013-203845
- Kuryata O, Lysunets T, Karavanska I, Semenov V. Duration till diagnosis and clinical profile of Sjogren's syndrome: data from real clinical practice in a single-center cohort. Egypt Rheumatol 2020;42:41-46. https://doi.org/10.1016/j.ejr.2019.05.003
- Mathews PM, Hahn S, Hessen M, et al. Ocular complications of primary Sjogren syndrome in men. Am J Ophthalmol 2015;160:447-452.e1. https://doi.org/10.1016/j.ajo.2015.06.004
- Fayyaz A, Kurien BT, Scofield RH. Autoantibodies in Sjogren's syndrome. Rheum Dis Clin North Am 2016;42:419-434. https://doi.org/10.1016/j.rdc.2016.03.002
- Baer AN, Gutierrez L, DeMarco MM, et al. Prevalence of seronegative Sjogren's syndrome: a comparative study from the Sjogren's International Collaborative Clinical Alliance (SICCA) cohort. Paper presented at: 2013 ACR/ARHP Annual Meeting; 2013 Oct 25-30; San Diego, USA. pp. S226.
- Varela-Centelles P, Seoane-Romero JM, Sanchez-Sanchez M, Gonzalez-Mosquera A, Diz-Dios P, Seoane J. Minor salivary gland biopsy in Sjogren's syndrome: a review and introduction of a new tool to ease the procedure. Med Oral Patol Oral Cir Bucal 2014;19:e20-e23.
- Kruszka P, O'Brian RJ. Diagnosis and management of Sjogren syndrome. Am Fam Physician 2009;79:465-470.
- Smedby KE, Hjalgrim H, Askling J, et al. Autoimmune and chronic inflammatory disorders and risk of non-Hodgkin lymphoma by subtype. J Natl Cancer Inst 2006;98:51-60. https://doi.org/10.1093/jnci/djj004
- Theander E, Vasaitis L, Baecklund E, et al. Lymphoid organisation in labial salivary gland biopsies is a possible predictor for the development of malignant lymphoma in primary Sjogren's syndrome. Ann Rheum Dis 2011;70:1363-1368. https://doi.org/10.1136/ard.2010.144782
- Risselada AP, Kruize AA, Goldschmeding R, Lafeber FP, Bijlsma JW, van Roon JA. The prognostic value of routinely performed minor salivary gland assessments in primary Sjogren's syndrome. Ann Rheum Dis 2014;73:1537-1540. https://doi.org/10.1136/annrheumdis-2013-204634
- Palm O, Garen T, Berge Enger T, et al. Clinical pulmonary involvement in primary Sjogren's syndrome: prevalence, quality of life and mortality--a retrospective study based on registry data. Rheumatology (Oxford) 2013;52:173-179. https://doi.org/10.1093/rheumatology/kes311
- Sjogren's Foundation. Breakthrough goal. Sjogren's Foundation accomplishes 5-year breakthrough goal [Internet]. Reston: Sjogren's Foundation; c2018 [cited 2021 May 4]. Available from: https://www.sjogrens.org/about-us/history/breakthrough-goal.