On-line First

J Rheum Dis

Published online June 12, 2024

© Korean College of Rheumatology

Clinical characteristics of chronic sclerosing sialadenitis as a distinctive entity from primary Sjögren’s syndrome

Eui-Jong Kwon , M.D., Youngjae Park , M.D., Ph.D., Seung-Ki Kwok , M.D., Ph.D., Ji Hyeon Ju , M.D., Ph.D.

Division of Rheumatology, Department of Internal Medicine, Seoul St. Mary’s Hospital, College of Medicine, The Catholic University of Korea, Seoul, Korea

Correspondence to : Ji Hyeon Ju, https://orcid.org/0000-0002-1381-5466
Division of Rheumatology, Department of Internal Medicine, Seoul St. Mary’s Hospital, College of Medicine, The Catholic University of Korea, 222 Banpo-daero, Seocho-gu, Seoul 06591, Korea. E-mail: juji@catholic.ac.kr

Received: April 5, 2024; Revised: May 15, 2024; Accepted: May 16, 2024

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract

Objective: This study aimed to elucidate the clinical and laboratory differences between chronic sclerosing sialadenitis (CSS) and primary Sjögren’s syndrome (pSS), highlighting CSS as a distinct pathological entity within the spectrum of salivary gland pathology.
Methods: This retrospective, single-center study was conducted at Seoul St. Mary’s Hospital between January 2000 and December 2022. Patients diagnosed with CSS via salivary gland biopsy were included, and those with IgG4-related disease (IgG4-RD) or other confounding factors were excluded. Clinical and laboratory CSS profiles were compared with those of a control group of patients with typical pSS from the Korean Initiative of Primary Sjögren’s Syndrome (KISS) prospective cohort study. Twenty-one with CSS and 501 patients with pSS from Seoul St. Mary’s Hospital were retrospectively analyzed.
Results: Patients with CSS were older at diagnosis, had a lower prevalence of ocular symptoms, and exhibited distinct immunological markers compared to those with pSS. Logistic regression analysis revealed that anti-Ro antibody positivity, elevated erythrocyte sedimentation rate levels, low serum complement 3 levels, and accompanying dry eye symptoms were factors distinguishing pSS from CSS.
Conclusion: Even after excluding IgG4-RD, CSS was significantly different from pSS in terms of clinical and laboratory findings. Recognition of these differences is crucial for the accurate diagnosis and management of CSS, underscoring its status as a distinct pathological entity among salivary gland pathologies.

Keywords Chronic sclerosing sialadenitis, Küttner’s tumor, Sjogren’s syndrome, Immunoglobulin G4-related disease

Article

On-line First

J Rheum Dis

Published online June 12, 2024

Copyright © Korean College of Rheumatology.

Clinical characteristics of chronic sclerosing sialadenitis as a distinctive entity from primary Sjögren’s syndrome

Eui-Jong Kwon , M.D., Youngjae Park , M.D., Ph.D., Seung-Ki Kwok , M.D., Ph.D., Ji Hyeon Ju , M.D., Ph.D.

Division of Rheumatology, Department of Internal Medicine, Seoul St. Mary’s Hospital, College of Medicine, The Catholic University of Korea, Seoul, Korea

Correspondence to:Ji Hyeon Ju, https://orcid.org/0000-0002-1381-5466
Division of Rheumatology, Department of Internal Medicine, Seoul St. Mary’s Hospital, College of Medicine, The Catholic University of Korea, 222 Banpo-daero, Seocho-gu, Seoul 06591, Korea. E-mail: juji@catholic.ac.kr

Received: April 5, 2024; Revised: May 15, 2024; Accepted: May 16, 2024

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract

Objective: This study aimed to elucidate the clinical and laboratory differences between chronic sclerosing sialadenitis (CSS) and primary Sjögren’s syndrome (pSS), highlighting CSS as a distinct pathological entity within the spectrum of salivary gland pathology.
Methods: This retrospective, single-center study was conducted at Seoul St. Mary’s Hospital between January 2000 and December 2022. Patients diagnosed with CSS via salivary gland biopsy were included, and those with IgG4-related disease (IgG4-RD) or other confounding factors were excluded. Clinical and laboratory CSS profiles were compared with those of a control group of patients with typical pSS from the Korean Initiative of Primary Sjögren’s Syndrome (KISS) prospective cohort study. Twenty-one with CSS and 501 patients with pSS from Seoul St. Mary’s Hospital were retrospectively analyzed.
Results: Patients with CSS were older at diagnosis, had a lower prevalence of ocular symptoms, and exhibited distinct immunological markers compared to those with pSS. Logistic regression analysis revealed that anti-Ro antibody positivity, elevated erythrocyte sedimentation rate levels, low serum complement 3 levels, and accompanying dry eye symptoms were factors distinguishing pSS from CSS.
Conclusion: Even after excluding IgG4-RD, CSS was significantly different from pSS in terms of clinical and laboratory findings. Recognition of these differences is crucial for the accurate diagnosis and management of CSS, underscoring its status as a distinct pathological entity among salivary gland pathologies.

Keywords: Chronic sclerosing sialadenitis, Kü,ttner’s tumor, Sjogren’s syndrome, Immunoglobulin G4-related disease

JRD
Jul 01, 2024 Vol.31 No.3, pp. 133~189
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