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Pomorska A, Świętoń D, Lieberman SM, et al. Recurrent or persistent salivary gland enlargement in children: When is it Sjögren's?. Semin Arthritis Rheum. 2021;doi: 10.1016/j.semarthrit.2021.11.011.
Pomorska, A., Świętoń, D., Lieberman, S. M., Bryl, E., Kosiak, W., Pęksa, R., Chorążewicz, J., Kochańska, B., Kowalska-Skabara, J., Szumera, M., Brzoznowski, W., Jaworski, R., & Irga-Jaworska, N. (2021). Recurrent or persistent salivary gland enlargement in children: When is it Sjögren's?. Seminars in arthritis and rheumatism, . https://doi.org/10.1016/j.semarthrit.2021.11.011
Pomorska, Anna, et al. "Recurrent or persistent salivary gland enlargement in children: When is it Sjögren's?." Seminars in arthritis and rheumatism vol. (2021). doi: https://doi.org/10.1016/j.semarthrit.2021.11.011
Pomorska A, Świętoń D, Lieberman SM, Bryl E, Kosiak W, Pęksa R, Chorążewicz J, Kochańska B, Kowalska-Skabara J, Szumera M, Brzoznowski W, Jaworski R, Irga-Jaworska N. Recurrent or persistent salivary gland enlargement in children: When is it Sjögren's?. Semin Arthritis Rheum. 2021 Dec 30; doi: 10.1016/j.semarthrit.2021.11.011. Epub 2021 Dec 30. PMID: 35000785.
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Semin Arthritis Rheum. 2021 Dec 30; doi: 10.1016/j.semarthrit.2021.11.011. Epub 2021 Dec 30.

Recurrent or persistent salivary gland enlargement in children: When is it Sjögren's?.

Seminars in arthritis and rheumatism

Anna Pomorska, Dominik Świętoń, Scott M Lieberman, Ewa Bryl, Wojciech Kosiak, Rafał Pęksa, Juliusz Chorążewicz, Barbara Kochańska, Justyna Kowalska-Skabara, Małgorzata Szumera, Wojciech Brzoznowski, Radosław Jaworski, Ninela Irga-Jaworska

Affiliations

  1. Department of Pediatrics, Hematology and Oncology, Medical University of Gda?sk, D?binki 7, Gda?sk 80-952, Poland. Electronic address: [email protected].
  2. 2nd Department of Radiology, Medical University of Gda?sk, Gda?sk, Poland.
  3. Division of Rheumatology, Allergy, and Immunology, Stead Family Department of Pediatrics, Carver College of Medicine, University of Iowa, Iowa, IA, United States.
  4. Department of Pathology and Experimental Rheumatology, Medical University of Gda?sk, Gda?sk, Poland.
  5. Department of Pediatrics, Hematology and Oncology, Medical University of Gda?sk, D?binki 7, Gda?sk 80-952, Poland.
  6. Pathology Department, Medical University of Gda?sk, Gda?sk, Poland.
  7. Department of Ophthalmology, Medical University of Gda?sk, Gda?sk, Poland.
  8. Department of Conservative Dentistry, Medical University of Gda?sk, Gda?sk, Poland.
  9. Department of Pediatrics, Gastroenterology, Hepatology and Nutrition, Medical University of Gda?sk, Gda?sk, Poland.
  10. Department of Otolaryngology, Medical University of Gda?sk, Gda?sk, Poland.
  11. Department of Pediatric Cardiothoracic Surgery, Children's Memorial Health Institute, Warsaw, Poland.

PMID: 35000785 DOI: 10.1016/j.semarthrit.2021.11.011

Abstract

OBJECTIVES: To describe characteristic features in children with recurrent or persistent salivary gland enlargement and to propose a diagnostic algorithm with specific consideration for Sjögren's disease (SD).

METHODS: In this single-center, prospective study, 45 patients < 18 years, with recurrent or persistent salivary gland enlargement of unknown etiology were enrolled from 2006 to 2019. We collected detailed clinical information to characterize this group of patients including specific details of their major salivary gland signs and symptoms. We compared clinical, laboratory and radiological parameters between 4 groups based on the results of labial salivary gland biopsy (LSGB) and between patients who met existing SD criteria or not.

RESULTS: 44 patients, with a mean age of 6.8 years and female to male ratio 21:23 were observed over a mean of 3.8 years. Characteristics of salivary gland swelling episodes varied considerably between individuals, but the majority experienced ≤5 episodes per year, lasting ≤ 1 week, with swelling affecting either or both glands. Ocular and oral dryness symptoms were observed only in 25% and 59% patients, respectively. The majority were positive for ANA, but negative for SD-specific antibodies. A total of 75% patients fulfilled at least one of the existing SD criteria.

CONCLUSION: SD is a major cause of recurrent salivary gland enlargement in children. For children meeting adult criteria, the diagnosis of SD is clear. However, for the many children without dryness symptoms, objective dryness, or SD-specific antibodies, further workup including a combination of salivary gland imaging and histopathological examination can help establish the diagnosis of SD.

Copyright © 2021 Elsevier Inc. All rights reserved.

Keywords: Childhood; Juvenile; Parotitis; Pediatric; Sjögren's disease; Sjögren's syndrome

Conflict of interest statement

Declaration of Competing Interest The authors declare no conflicts of interest.

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