Abstract
Major salivary gland ultrasonography (SGUS) is increasingly being recognized as having critical roles in differentiating primary Sjögren’s syndrome (pSS) from other connective tissue disorders. Contrast-enhanced ultrasonography (CEUS) has been reported to evaluate microvascularity of lesions in different tissues with objective angiographic index, eliminating the observer-dependent defect of ultrasonography. However, there are few relevant studies concentrating on the application of CEUS in the diagnosis and assessment for pSS, and their clinical utility prospect remains uncertain. In this study, a total of 227 eligible patients were enrolled, including 161 pSS and 66 non-pSS patients with comprehensive ultrasonographic evaluation of the parotid and submandibular glands, including grayscale ultrasonography, color Doppler sonography (CDS), and CEUS. Compared with non-pSS, pSS patients had significantly higher grayscale ultrasound (US) scores and CDS blood grades in the parotid gland and significantly higher grayscale US and CEUS scores in the submandibular glands. Diagnostic model combining ultrasonographic signatures, anti-SSA/Ro60, and keratoconjunctivitis sicca (KCS) tests showed a remarkable discrimination [mean area under the curve (AUC)0.963 in submandibular glands and 0.934 in parotid glands] for pSS, and the nomogram provided excellent prediction accuracy and good calibration in individualized prediction of pSS. A combination of multiple ultrasonographical examinations of the major salivary glands (SGs) is a promising technique that may be used as a practical alternative to minor SG biopsy in the detection of pSS.
Highlights
Primary Sjögren’s syndrome is a common chronic systemic autoimmune disease characterized by focal lymphocytic infiltration of the exocrine glands, especially in the salivary and tear glands, mainly presenting as xerostomia, xerophthalmia, and other extraglandular involvements [1]
These patients were subsequently divided into 161 primary Sjögren’s syndrome (pSS) and 66 non-pSS subgroups based on the 2016 American College of Rheumatology (ACR)/European League Against Rheumatism (EULAR) [6] or 2012 ACR criteria [5]
We revealed that ultrasonographic signatures manifested a high discriminatory capability in distinguishing pSS from non-pSS, with a mean area under the curve (AUC) value of 0.807 for grayscale US scores and 0.764 for Contrast-enhanced ultrasonography (CEUS) scores
Summary
Primary Sjögren’s syndrome (pSS) is a common chronic systemic autoimmune disease characterized by focal lymphocytic infiltration of the exocrine glands, especially in the salivary and tear glands, mainly presenting as xerostomia, xerophthalmia, and other extraglandular involvements [1]. With a population prevalence of 0.05%–0.6% and a nearly 14:1 female/male ratio, pSS mostly affects middle-aged females between 30 and 50 years at the time of diagnosis [2, 3]. Detection of pSS is a challenge due to the complex nature and heterogeneity of the disease and no gold standard test. Current classification criteria [5, 6] have greatly improved this situation, with histopathology of minor salivary gland biopsy (MSGB) a dominant factor. The invasive nature of MSGB, with high risks of several complications such as persistent sensory deprivation caused by local nerve injury and pyogenic granuloma [7, 8], means it is still not accepted by all patients. There is an urgent unmet need for improved and non-invasive methods to facilitate the detection of pSS
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