Intraoperative Visualization and Treatment of Salivary Gland Dysfunction in Sjögren's Syndrome Patients Using Contrast-Enhanced Ultrasound Sialendoscopy (CEUSS)

Abstract

In sialendoscopy, ducts are dilated and the salivary glands are irrigated with saline. Contrast-enhanced ultrasound sialendoscopy (CEUSS), using microbubbles, may facilitate the monitoring of irrigation solution penetration in the ductal system and parenchyma. It is imperative to test CEUSS for its safety and feasibility in Sjögren's syndrome (SS) patients. CEUSS was performed on 10 SS patients. The primary outcomes were safety, determined by the occurrence of (serious) adverse events ((S)AEs), and feasibility. The secondary outcomes were unstimulated and stimulated whole saliva (UWS and SWS) flow rates, xerostomia inventory (XI), clinical oral dryness score, pain, EULAR Sjögren's syndrome patient reported index (ESSPRI), and gland topographical alterations. CEUSS was technically feasible in all patients. Neither SAEs nor systemic reactions related to the procedure were observed. The main AEs were postoperative pain (two patients) and swelling (two patients). Eight weeks after CEUSS, the median UWS and SWS flow had increased significantly from 0.10 to 0.22 mL/min (p = 0.028) and 0.41 to 0.61 mL/min (p = 0.047), respectively. Sixteen weeks after CEUSS, the mean XI was reduced from 45.2 to 34.2 (p = 0.02). We conclude that CEUSS is a safe and feasible treatment for SS patients. It has the potential to increase salivary secretion and reduce xerostomia, but this needs further investigation.

Keywords: Sjogren’s syndrome; Sjögren’s syndrome; endoscopy; microbubbles; saliva; sialendoscopy; ultrasound; xerostomia.

Figure 1

Flow diagram of the allocation of participants.

Figure 2

Images of (A) left parotid gland with complete homogeneous dense penetration of microbubbles. (B) Right submandibular gland (SMG) with complete, inhomogeneous dense penetration of microbubbles. (C) Right parotid gland with complete homogenous penetration of microbubbles with moderate density. (D) Left SMG with no penetration of microbubbles. Only the main duct is visible. Yellow areas: microbubbles; Arrow: Wharton’s duct. Please note: the images are from four different patients.