Abstract
Background: Sicca syndrome represents a heterogeneous group of conditions, such as Sjögren syndrome, causing xerophthalmiaand xerostomia. This study characterizes in depth patients with Sicca syndrome and evaluates salivary gland ultrasound (SGUS).Methods: Principal component analysis and hierarchical clustering of clinical parameters, such as ESSPRI, ESSDAI and laboratory data, were performed on all referrals for assessment of Sicca symptoms between October 2018 and March 2021. SGUS and labial gland biopsies were compared across groups.Results: A total of 583 patients were assessed. Objective dryness was confirmed in 73% of the patients. Cluster analysis identified 3 groups with post-hoc analysis confirming distinct phenotypes: Somatic Group (283/583; 49%) with more frequent symptoms but limited objective dryness; Dry Without Autoimmune Features (DAFneg, 206/584; 35%), and Dry With Autoimmune Features (DAFpos, 94/584;16%). DAFpos patients had highest autoantibody titers (anti-SSA(Ro) 240 vs. 3.6 vs. 3.8; p < 0.001), most extra-glandular manifestations (p < 0.001), and highest median SGUS Score (DAFpos: 8 [IQR 4–10] vs. SG: 2 [1–4] vs. DAFneg 4 [2–5]; p < 0.001). No tangible correlation with primary Sjögren syndrome criteria was observed.Discussion: SGUS score correlated with a subset of patients with Sjögren syndrome, identified in the DAFpos cluster. This study highlights heterogeneity within sicca and, indeed, Sjögren syndrome, highlighting the need for further studies.
Highlights
Sicca syndrome represents a heterogeneous group of conditions, such as Sjögren syndrome, causing xerophthalmiaand xerostomia
Hierarchical clustering identified three clearly demarcated groups (Figure 1), which were phenotypically characterized in detail (Table 1)
This study evaluated the utility of Salivary gland ultrasound (SGUS) in unselected patients referred for evaluation of Sjögren syndrome, and explored the relationship between Sicca syndrome and Sjögren syndrome beyond the American College of Rheumatology (ACR)/European League against Rheumatism (EULAR) criteria
Summary
Sicca syndrome represents a heterogeneous group of conditions, such as Sjögren syndrome, causing xerophthalmiaand xerostomia. PSS requires evidence of autoimmune inflammation of salivary or lacrimal glands, as outlined in the joint American College of Rheumatology (ACR) and European League against Rheumatism (EULAR) classification criteria [3]. These are deliberately broad, affording some heterogeneity in clinical features, but require either the presence of anti-SSA antibodies or evidence of lymphocytic sialadenitis on labial gland biopsy (LBx). Salivary gland ultrasound (SGUS) has been purported as a low-cost and radiation-free alternative for many years, with hypoechoic lesions correlating with more severe disease in a variety of scoring systems [6,7,8]. Specificity has been favorable, reported sensitivity has been moderate [10]
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