The Prevalence of Sjögren's Disease in Dental Clinics in the Netherlands Compared with the Prevalence in a Systematic Literature Review of Studies in Other Countries.

Background/Objectives: Sjögren's disease (SjD) is an autoimmune disease causing irreversible damage to the exocrine glands but can have symptoms throughout the entire body. The aim of this study is to determine the prevalence of Sjogren's disease (SjD) in the Netherlands, compare this with the prevalence for other countries in a systematic literature review. Methods: In the first part of this study, the prevalence of SjD was determined at two academic dental clinics in the Netherlands by electronically analysing patient records. In the second part of this study, a systematic literature search was performed in PubMed. Studies in the English language reporting prevalence ratios (PRs), incidence ratios (IRs) or sufficient data to calculate these parameters were included. Population-based studies and population surveys aiming to examine an entire geographic region or using a clearly defined sampling procedure were included. Review studies were excluded. Studies that did not report sufficient data or contained no original data were excluded. Included studies were assessed using the Newcastle-Ottawa assessment scale. Results: At the dental clinic in Amsterdam, 76 SJD patients were identified among a patient population of 81941, resulting in a prevalence ratio of 93 per 100,000 (0.093%) patients. In Nijmegen, 21 SjD patients were identified in a total patient population of 14,240, resulting in a prevalence ratio of 147 per 100,000 (0.15%). Thirty-one studies were included in the systematic review. They varied in diagnostic criteria for SjD with the American-European Consensus Group (AECG) criteria being the most widely used. The reported prevalence ratio varied from 0.008% to 3.3%. The overall pooled prevalence ratio of SjD using the AECG criteria was 0.031%, while the pooled prevalence of SjD using the EU criteria was 0.029%. The overall pooled incidence ratio was 5.2 (95%CI 4.7 to 5.6) per 100,000 person-years. Conclusions: The estimated prevalence ratio of SjD in the Netherlands (0.09% to 0.15%) falls within the worldwide range but is higher than the worldwide pooled prevalence ratio.

The minimally important difference for the Xerostomia Inventory among Sjögren's disease patients.

OBJECTIVE: Until now, the clinically relevant improvement for the Xerostomia Inventory (XI) has not been defined. Therefore, our aim was to determine the Minimally Important Difference (MID) of the XI for improvement in dry-mouth symptoms in SjD patients. METHOD: The study recruited 34 SjD patients who underwent sialendoscopy of major salivary glands and 15 SjD patients in a nonintervention control group. XI scores were assessed at several time points. The MID was determined from the mean difference in XI scores between the groups with and without improvement. RESULTS: In the control group, no significant XI score changes were seen. In the sialendoscopy group, a clinically relevant XI score change of four scale points was identified after 1 week. For a prolonged duration (≥16 weeks), a minimum reduction of seven scale points in the XI score was required to indicate clinically relevant improvement. CONCLUSION: In SjD patients, a minimum change of four points in the XI score indicates a clinically relevant improvement for evaluating short-term effects. For prolonged effects, a clinically relevant improvement requires a MID of seven points. The determination of the MID in XI could assist in future studies that evaluate changes in xerostomia.

Implant supported overdentures in Sjögren's disease patients: a multicentre prospective cohort study.

OBJECTIVES: To prospectively investigate patient-reported outcomes and clinical performance of implant supported overdentures in edentulous Sjögren's disease (SjD) patients compared to subjects without SjD. METHODS: 51 implants were placed in 12 patients with SjD and 50 implants in 12 non-SjD patients to support overdentures. Clinical performance, marginal bone-level changes, patient satisfaction and oral health related quality of life (OHRQoL) were assessed at 1 (T1), 6 (T6), 12 (T12) and 18 (T18) months after placement of the overdenture. Patient satisfaction, ability to chew and OHRQoL were assessed with validated questionnaires. Marginal bone-level changes were measured on standardised dental radiographs. Clinical parameters included implant and overdenture survival, plaque, bleeding and gingival indices, and probing depth. RESULTS: OHRQoL in patients with SjD improved significantly after placement of implant supported overdentures at all measuring moments compared to baseline (p<0.05). Nevertheless, ability to chew tough and hard food was significantly better for non-SjD patients at all timepoints after placement of an implant supported overdenture (p<0.05). Implant survival at T18 was 100% in the patients with SjD and 98% in the non-SS group. Mean marginal bone loss at T18 did not differ between patients with SjD and non-SS patients, 1.12±0.74 mm and 1.43±1.66 mm, respectively (p=0.58). Clinical performance was good with no differences between the groups for all outcome measures (p>0.05). CONCLUSIONS: Implant-supported overdentures have a positive effect on OHRQoL and dental implants can be successfully applied in edentulous patients with SjD with nearly similar outcomes as in non-SjD subjects.

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

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.

Dental implants in dentate primary and secondary Sjögren's syndrome patients: A multicenter prospective cohort study.

OBJECTIVES: To prospectively assess the clinical performance and patient-reported outcomes of dental implants in dentate patients with primary and secondary Sjögren's syndrome (pSS and sSS, respectively) compared to patients without SS. MATERIALS AND METHODS: Thirty-seven implants were placed in 17 patients with pSS/sSS and 26 implants in 17 non-SS patients to replace missing (pre)molars. Clinical performance, marginal bone-level changes, patient satisfaction, and oral health-related quality of life (OHRQoL) were assessed at 1 (T1), 6 (T6), 12 (T12), and 18 (T18) months after placement of the superstructure. Marginal bone-level changes were measured on standardized dental radiographs. Clinical parameters included implant and crown survival, plaque, bleeding and gingival indices, and probing depth. Patient satisfaction and OHRQoL were assessed with validated questionnaires. RESULTS: Implant survival at T18 was 100% in the patients with pSS/sSS and 96.2% in the non-SS group. Mean marginal bone loss at T18 did not differ between patients with pSS/sSS and non-SS patients, 1.10 ± 1.04 and 1.04 ± 0.75 mm, respectively (p = .87). Clinical performance was good with no differences between the groups for all outcome measures (p > .05). OHRQoL in patients with pSS/sSS had improved significantly after placement of implant supported crowns at all measuring moments compared to baseline (p < .05). Nevertheless, patient satisfaction and OHRQoL remained significantly higher for patients without SS at all measuring moments (p < .05). CONCLUSION: Dental implants can be successfully applied in dentate patients with pSS/sSS and have a positive effect on OHRQoL.

Intraoperative visualisation and treatment of salivary glands in Sjögren's syndrome by contrast-enhanced ultrasound sialendoscopy (CEUSS): protocol for a phase I single-centre, single-arm, exploratory study.

INTRODUCTION: We established a promising sialendoscopic treatment for in vivo enhancement of salivation in salivary glands affected by Sjögren's syndrome (SS). In this technique, the ducts of the salivary glands are irrigated with saline and steroids. This allows for dilatation of ductal strictures and removal of debris. Unfortunately, it is not possible to assess the delivery and penetration of saline or medications in the ductal system and parenchyma. To address this problem, we will conduct contrast-enhanced ultrasound sialendoscopy (CEUSS) using sulphur hexafluoride microbubbles. To the best of our knowledge, microbubbles have never been used for the treatment of salivary glands in SS. It is, therefore, imperative to test this application for its safety and feasibility. METHODS AND ANALYSIS: A single-arm phase I study will be performed in 10 SS patients. Under local anaesthesia, ultrasound (US) guided infusion of the parotid and submandibular glands with microbubbles will be performed. Continuous US imaging will be used to visualise the glands, including the location of strictures and occlusions. Main outcomes will be the evaluation of safety and technical feasibility of the experimental treatment. Secondary outcomes will consist of determinations of unstimulated whole mouth saliva flow, stimulated whole mouth saliva flow, stimulated parotid saliva flow, clinical oral dryness, reported pain, xerostomia, disease activity, salivary cytokine profiles and clinical SS symptoms. Finally, salivary gland topographical alterations will be evaluated by US. ETHICS AND DISSEMINATION: Ethical approval for this study was obtained from the Medical Ethics Committee of the Amsterdam University Medical Centre, Amsterdam, The Netherlands (NL68283.029.19). data will be presented at national and international conferences and published in a peer-reviewed journal. The study will be implemented and reported in line with the Standard Protocol Items: Recommendations for Interventional Trials' statement. TRIAL REGISTRATION NUMBERS: The Netherlands Trial Register: NL7731, MREC Trial Register: NL68283.029.19; Pre-results.

Sialendoscopy enhances salivary gland function in Sjögren's syndrome: a 6-month follow-up, randomised and controlled, single blind study.

OBJECTIVES: To assess the effect of sialendoscopy of the major salivary glands on salivary flow and xerostomia in patients with Sjögren's syndrome (SS). METHODS: Forty-nine patients with SS were randomly assigned to a control group (n=15) and two intervention groups: irrigation of the major glands with saline (n=16) or with saline followed by triamcinolone acetonide (TA) in saline (n=18). Unstimulated whole saliva flow (UWS), chewing-stimulated whole saliva flow (SWS), citric acid-stimulated parotid flow (SPF), Clinical Oral Dryness Score (CODS), Xerostomia Inventory (XI) score and the European League Against Rheumatism (EULAR) SS Patient-Reported Index (ESSPRI) were obtained 1 week (T0) before, and 1 (T1), 8 (T8), 16 (T16) and 24 (T24) weeks after sialendoscopy. RESULTS: Median baseline UWS, SWS and SPF scores were 0.14, 0.46 and 0.22 mL/min, respectively. After intervention, significant increases in UWS and SWS were observed in the saline group (at T8 (P=0.013) and T24 (P=0.004)) and the saline/TA group (at T24 (P=0.03) and T=16 (P=0.035)). SPF was increased significantly in the saline/TA group at T24 (P=0.03). XI scores declined after sialendoscopy in both intervention groups. Compared with the control group, CODS, XI and ESSPRI improved in the intervention groups. UWS, SWS and SPF were higher in the intervention groups compared with the control group, but these differences were not significant except for SPF in the saline/TA group at T24 (P=0.005). CONCLUSIONS: Irrigation of the major salivary glands in patients with SS enhances salivary flow and reduces xerostomia up to 6 months after sialendoscopy.