Bilateral Parotid Gland Cysts as an Atypical Indicator of Sjögren's Syndrome: A Case Study and Literature Overview

Document Type : Case Report

Authors

Maxillofacial Surgery Unit, Department of Neurosciences, Reproductive and Odontostomatological Sciences, University of Naples Federico II, Naples, Italy.

10.22038/ijorl.2025.85419.3869

Abstract

Introduction:
Sjögren's syndrome is a systemic autoimmune disorder that gradually impairs exocrine function, primarily affecting lacrimal and salivary glands.
Case Report:
We describe an unusual presentation involving an elderly female patient diagnosed with late-stage Sjögren's syndrome. Laboratory testing eliminated viral infections including HIV and HCV. Fine-needle aspiration biopsy of parotid swellings revealed inflammatory cystic lesions, excluding malignancy and other common cystic conditions. MRI revealed several fluid-filled nodules in both glands.
Conclusion:
This report supports including Sjögren's syndrome in the differential diagnosis for bilateral cystic lesions of the parotid glands. A synthesis of similar literature cases is included.

Keywords

Main Subjects

References

  1. Vivino FB, Carsons SE, Foulks G, Daniels TE, Parke A, Brennan MT, Forstot SL, Scofield RH, Hammitt KM. New Treatment Guidelines for Sjögren's Disease. Rheum Dis Clin North Am. 2016 Aug;42(3):531-51. doi: 10.1016/j.rdc.2016.03.010. PMID: 27431353; PMCID: PMC5812283.
  2. Brito-Zerón P, Baldini C, Bootsma H, Bowman SJ, Jonsson R, Mariette X, Sivils K, Theander E, Tzioufas A, Ramos-Casals M. Sjögren’s Nat Rev Dis Primers. 2016 Jul 7;2:16047. doi: 10.1038/nrdp.2016.47. PMID: 27383445.
  3. Committeri U, Barone S, Salzano G, Arena A, Borriello G, Giovacchini F, Fusco R, Vaira LA, Scarpa A, Abbate V, Ugga L, Piombino P, Ionna F, Califano L, Orabona GD. Support Tools in the Differential Diagnosis of Salivary Gland Tumors through Inflammatory Biomarkers and Radiomics Metrics: A Preliminary Study. Cancers (Basel). 2023 Mar 21;15(6):1876. doi: 10.3390/ cancers 15061876. PMID: 36980760; PMCID: PMC1004 7378.
  4. Abbate V, Barone S, Troise S, Laface C, Bonavolontà P, Pacella D, Salzano G, Iaconetta G, Califano L, Dell'Aversana Orabona G. The Combination of Inflammatory Biomarkers as Prognostic Indicator in Salivary Gland Malignancy. Cancers (Basel). 2022 Nov 30;14(23):5934. doi: 10.3390/cancers14235934. PMID: 36497416; PMCID: PMC9740974.
  5. Abbate V, Dell’Aversana Orabona G, Barone S, Troise S, Bonavolontà P, Pacella D, et al. Relevance of inflammatory biomarkers in salivary gland cancers management. Eurasian J Med Oncol. 2021;5(4):311–317. doi:10.14744/ejmo.2021.85983
  6. Abbate V, Barone S, Borriello G, Troise S, Bonavolontà P, Pacella D, Vaira LA, Turri-Zanoni M, Cuéllar CN, Califano L, Dell' Aversana Orabona G. Diagnostic performance of inflammatory biomarkers and cytological analysis in salivary gland tumors. Head Neck. 2023 Dec;45(12):3015-3023. doi: 10.1002/hed.27528. Epub 2023 Sep 26. PMID: 37752706.
  7. Zhurakivska K, Maiorano E, Nocini R, et al. Necrotizing sialometaplasia can hide the presence of salivary gland tumors: A case series. Oral Dis. 2019;25(4):1084-1090. doi:10.1111/odi.13066
  8. Provost TT. Anti-Ro(SSA) and anti-La(SSB) antibodies in lupus erythematosus and Sjögren's syndrome. Keio J Med. 1991 Jun;40(2):72-7. doi: 10.2302/kjm.40.72. PMID: 1881026.
  9. Plaza G, Domínguez MP, Bueno A. Bilateral parotid cysts as presentation of Sjögren's syndrome. J Laryngol Otol. 2003 Feb;117(2):151-2. doi: 10.1258/002221503762624675. PMID: 12625896.
  10. Seo BF, Ju RK, Kwok SK, Oh DY. Unusual Sjögren's Syndrome with Bilateral Parotid Cysts. Arch Craniofac Surg. 2014 Aug;15(2):98-101. doi: 10.7181/acfs.2014.15.2.98. Epub 2014 Aug 14. PMID: 28913199; PMCID: PMC5556822.
  11. Toh AS, Broomfield SJ, Teh LS, Aslam MB, Duncan G, Morar P. Bilateral multicystic parotid masses in primary Sjögren’s Ear Nose Throat J. 2011 Oct;90(10):E20-2. doi: 10.1177/ 014556131109001021. PMID: 22033967.
  12. Ahmad I, Ray J, Cullen RJ, Shortridge RT. Bilateral and multicystic major salivary gland disease: a rare presentation of primary Sjögren's syndrome. J Laryngol Otol. 1998 Dec; 112(12): 1196-8. doi: 10.1017/s0022215100142847. PMID: 10209623.
  13. Skinner J, Fowler J, Park J, You P. Paediatric Sjögren's Syndrome With Bilateral Parotid Cysts: A Case Report. Cureus. 2023 Aug 6;15(8):e43033. doi: 10.7759/cureus.43033. PMID: 37674949; PMCID: PMC10479929.
  14. Janus JR, Mzaik O, Bhatt AA. Parotid sebaceous lymphadenoma associated with Sjogren’s syndrome: Review of pathologic and radiologic characteristics and clinical presentation. J Clin Imaging Sci 2022;12:14. doi:10. 25259/ JCIS_ 14_2022
  15. Gadodia A, Seith A, Sharma R. Unusual presentation of Sjögren’s syndrome: multiple parotid cysts. Ear Nose Throat J. 2012 Nov; 91(11): E17-9. PMID: 23288799.
  16. Som PM, Brandwein MS, Silvers A. Nodal inclusion cysts of the parotid gland and parapharyngeal space: a discussion of lymphoepithelial, AIDS-related parotid, and branchial cysts, cystic Warthin's tumors, and cysts in Sjögren's syndrome. Laryngoscope. 1995 Oct; 105(10): 1122-8. doi: 10.1288/00005537-1995 10000-00020. PMID: 7564846.
Iranian Journal of Otorhinolaryngology
Volume 37, Issue 6
November and December 2025
Pages 349-352

Files

Share

How to cite

Statistics