Ultrasonographic characteristics of major salivary glands in anti-centromere antibody-positive primary Sjögren's syndrome.

Hong Ki Min,Kyung-Ann Lee,Seung-Ki Kwok,Sang-Heon Lee,Se-Hee Kim,Youngjae Park,Hae-Rim Kim,Masataka Kuwana

doi:10.1371/journal.pone.0259519

Abstract

To investigate salivary gland ultrasonography (SGUS) findings in primary Sjögren's syndrome (pSS) patients positive for the anti-centromere antibody (ACA) and compare these with those in ACA-negative pSS patients. We analyzed demographic, clinical, laboratory, and SGUS data of pSS patients who fulfilled the 2002 American-European Consensus Group classification criteria for pSS. SGUS findings of four major salivary glands (bilateral parotid and submandibular glands) were scored in five categories and compared between ACA-positive and ACA-negative pSS patients. Linear regression analysis was performed to elucidate the factors associated with SGUS score. In total, 121 pSS patients were enrolled (19, ACA-positive). The ACA-positive patients were older (67.0 vs 58.0 years, P = 0.028), whereas anti-Ro/SSA and anti-La/SSB positivity was more prevalent in the ACA-negative group (89.2% vs 21.1%, P < 0.001, and 47.1% vs 10.5%, P = 0.007, respectively). The total SGUS and hypoechoic area scores were lower in ACA-positive patients (16.0 vs 23.0, P = 0.027, and 4.0 vs 7.0, P = 0.004, respectively). In univariate regression analysis, being positive for unstimulated salivary flow rate (USFR < 1.5 ml/15 min), anti-Ro/SSA, and rheumatoid factor were positively associated whereas ACA positivity was negatively associated with the SGUS score. In multivariate regression analysis, being positive for USFR, anti-Ro/SSA, and rheumatoid factor showed significant association with the SGUS score. ACA-positive pSS patients showed a lower SGUS score than ACA-negative patients, which was especially prominent in the hypoechoic area component.

Highlights

Primary Sjogren’s syndrome is a systemic autoimmune disease which primarily affects exocrine glands and causes sicca symptoms
The total salivary gland ultrasonography (SGUS) and hypoechoic area scores were lower in anti-centromere antibody (ACA)-positive patients (16.0 vs 23.0, P = 0.027, and 4.0 vs 7.0, P = 0.004, respectively)
In univariate regression analysis, being positive for unstimulated salivary flow rate (USFR < 1.5 ml/15 min), anti-Ro/SSA, and rheumatoid factor were positively associated whereas ACA positivity was negatively associated with the SGUS score

Summary

Introduction

Primary Sjogren’s syndrome (pSS) is a systemic autoimmune disease which primarily affects exocrine glands and causes sicca symptoms. Major salivary gland ultrasonography (SGUS) has been suggested as an adjuvant tool for pSS diagnosis and shows a similar diagnostic power to sialoscintigraphy or minor salivary gland biopsy [9]. Since SGUS has the advantage of finding structural changes in the salivary gland of pSS patients, it has been suggested as an early diagnostic tool [11]. A recent study has shown that hyperechoic foci found during SGUS correlates with USFR [13], which suggests that SGUS findings could predict the structural changes in salivary glands, and the functional and histologic changes. The SGUS showed significant association with disease activity measured by European League Against Rheumatism Sjogren’s syndrome disease activity index, which imply that SGUS is useful for diagnosis of pSS and for predicting clinical activity of pSS [14]

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Results

Conclusion