A Novel Approach for Immediate Implant-Based Oral Rehabilitation in a Sjögren's Syndrome Patient Using Virtual Surgical and Prosthetic Planning.

Patients with Sjögren syndrome (SS) experience difficulties in wearing conventional dentures. After removal of all teeth, the oral rehabilitation is challenging and time consuming using conventional treatment protocols. Although implant-retained overdentures are beneficial for this specific patient group, the average total oral rehabilitation time (TORT) usually takes at least 9 months and needs to be reduced to increase patients' quality of life (QoL). In this paper, we report on a new treatment concept for immediate implant-based oral rehabilitation in a 77-year-old patient with partial edentulous SS. Because of persistent pain, discomfort, and retention problems with the conventional prosthetic devices, full clearance of the remaining mandibular dentition and immediate oral rehabilitation with an implant-retained overdenture were suggested. The treatment protocol included virtual surgical planning (VSP), combining a guided bone reduction of the mandibular alveolar process, immediate dental implant placement, and restoration using a prefabricated bar and placement of the overdenture. This method demands the use of ionizing 3D imaging optionally combined with an optical dental scan or a conventional impression. Furthermore, one needs to gain experience using VSP software. This novel treatment concept for immediate implant-based oral rehabilitation using VSP proved to be feasible and safe in a patient with SS, resulting in a significantly reduced TORT and improved QoL. Further research is needed to what extent this treatment concept could be beneficial to other patient groups, such as patients with head and neck cancer.

Sialendoscopy and Sjogren's Disease: A Systematic Review.

OBJECTIVES/HYPOTHESIS: This study is a systematic review of the literature which seeks to estimate the expected treatment outcomes of a patient with Sjogren's syndrome (SS) undergoing therapeutic sialendoscopy. STUDY DESIGN: Systematic Review. METHODS: PubMed, Scopus, and Cochrane library databases were used to search for studies published as of August 2020 regarding the treatment outcomes of SS with sialendoscopy. The key search terms included "Sjogren's syndrome" and "sialendoscopy." Only studies in the English language involving more than one human patient were included. PRISMA guidelines were followed in study inclusion and data extraction. The primary outcome assessed was improvement in patient symptoms. RESULTS: Six studies met criteria and were analyzed in this review, including 125 patients undergoing sialendoscopy of parotid and/or submandibular glands as well as 25 controls. Of these patients, 90% were female with an age range of 18 to 79 years. There was significant diversity in outcome reporting tools. The outcomes of symptom improvement were pooled qualitatively based on improvement noted in each study. Outcomes were defined as partial improvement if the measured outcomes improved and complete improvement if measured outcomes resolved entirely. Despite the limited number of studies on this topic, this meta-analysis suggests that a similar study of therapeutic sialendoscopy could expect to provide at least temporary improvement of symptoms 90% to 99% of the time. CONCLUSIONS: This review provides support for the application of sialendoscopy in the treatment of SS salivary disease. Larger studies with consistent outcome reporting tools and control groups are needed to validate these results and provide a consistent therapy protocol. Laryngoscope, 131:1474-1481, 2021.

Mesenchymal Stem Cell Enhances the Function of MDSCs in Experimental Sjögren Syndrome.

Primary Sjögren's syndrome (pSS) is a progressive systemic autoimmune disease characterized by lymphocytic infiltrates in exocrine glands, leading to the injury of salivary and lachrymal glands. Mesenchymal stem cells (MSCs) have been demonstrated to exert great potential in the treatment of various autoimmune diseases. Although MSCs have provide an effective therapeutic approach for SS treatment, the underlying mechanisms are still elusive. Our previous study has shown the reduced suppressive capacity of myeloid-derived suppressor cells (MDSCs) advanced the progression of experimental Sjögren's syndrome (ESS). In this study, we found that BM-MSCs significantly enhanced the suppressive function of MDSCs with high levels of Arginase and NO, decreased the levels of CD40, CD80, CD86, and MHC-II expression on MDSCs, thus attenuating the disease progression in ESS mice. Furthermore, the enhanced suppressive function of MDSCs was mediated by BM-MSC-secreted TGF-ß, and the therapeutic effect of BM-MSCs in inhibiting ESS was almost abolished after silencing TGF-ß in BM-MSCs. Taken together, our results demonstrated that BM-MSCs alleviated the ESS progression by up-regulating the immunosuppressive effect of MDSCs through TGF-ß/Smad pathway, offering a novel mechanism for MSCs in the treatment of pSS.

Diagnostic and treatment effects of sialendoscopy for patients with swelling of the parotid gland when sialoliths are undetected with computed tomography.

Between August 2009 and May 2016, 74 patients underwent sialoendoscopic surgery. 32 patients had parotid gland disease and 9 patients had intermittent swelling of the parotid gland and sialoliths were not detected with CT imaging. 4 patients were diagnosed with idiopathic Stensen's duct stenosis. Sialendoscopy directly confirmed Stensen's duct stenosis in 2 patients. However, the sialendoscope was unable to be inserted in the other 2 patients, who had stenosis of the orifice of the Stensen's duct. Balloon expansion of the duct was performed in these 2 patients and a steroid drug was injected into the duct in one patient. Complete remission was archived in one patient treated with sialendoscopy. Three patients had sialolithiasis. Microsialoliths and/or white floating matter was observed and removed using sialendoscopy. All patients experienced complete remission. In cases of Sjögren syndrome and recurrent parotitis, sialendoscopic surgery was performed, but the symptoms showed no improvement. For patients with microsialoliths, sialendoscopy may be most useful for diagnosis and treatment when the sialoliths are not detected with CT imaging. At present, sialendoscopic surgery have limitation in the treatment of Stensen's duct stenosis and may similarly have limitation in the treatment of Sjögren's syndrome and recurrent parotitis.

Complications of Sialendoscopy in Patients With Sjögren Syndrome.

PURPOSE: Recent literature suggests that sialendoscopy of the major salivary glands could alleviate symptoms of Sjögren syndrome (SS) and restore salivary function. The aim of this study was to evaluate the authors' experience of sialendoscopy of the salivary glands in patients with SS. MATERIALS AND METHODS: In this retrospective case series study, the surgical data of patients with SS who had undergone sialendoscopy at the VU University Medical Center (Amsterdam, The Netherlands) from November 2014 through April 2015 were used. Outcome measurements were successful entry to the salivary gland and completion of sialendoscopy as planned. Furthermore, pre- and postoperative complications were scored. Descriptive analysis of the data was performed. RESULTS: Surgical data of sialoendoscopic procedures in 26 patients with SS (24 women and 2 men; mean age, 57 yr; range, 27 to 72 yr) were analyzed. Sialendoscopy was successfully performed in 78 of 104 salivary glands (75%; 50 parotid and 28 submandibular glands) in the 26 patients. Sialendoscopy failed in 26 of the 104 sialoendoscopic procedures (25%; 2 parotid and 24 submandibular glands). In 16 salivary glands, the ductal orifice could not be identified. In 7 salivary glands, sialendoscopy could not be performed because of partial or complete stenosis of the salivary duct. In 3 salivary glands, sialendoscopy was not performed because of a ductal perforation. Three patients developed a postoperative infection. CONCLUSION: The overall rate of complications was limited and the sialoendoscopic complications in patients with SS could be regarded as minor. Most complications were seen for sialendoscopy of the submandibular glands in this specific patient category. Careful preoperative selection of patients and salivary glands could contribute to a lower rate of complications and more predictable results.

Sialendoscopic management of autoimmune sialadenitis: a review of literature.

Autoimmune diseases of major salivary glands include Sjögren's syndrome and a complex of disorders classified as immunoglobulin G4-related diseases. These pathologies are characterised by an autoimmune reaction mediated by T-helper lymphocytes that targets the ducts of exocrine glands in Sjögren's syndrome and glandular parenchyma in immunoglobulin G4-related diseases. Immunoglobulin G4-related diseases represent recently introduced multi-organ diseases that also involve the salivary glands. However, the morbid conditions once known as Mikulicz's disease and Kuttner's tumour were recently considered as two variants of immunoglobulin G4-related diseases affecting the major salivary glands ( immunoglobulin G4-related sialadenitis). This review briefly summarises the pathogenesis and clinical features of autoimmune diseases of the major salivary glands, focusing on the diagnostic and therapeutic role of sialendoscopy.

Spontaneous Canalicular Plug Erosion After Punctal Plug Placement.

PURPOSE: To describe a case of keratoconjunctivitis sicca treated with punctal plug placement with the first reported complication of subsequent plug erosion through the canaliculus and extrusion through the lid. METHODS: Punctal plugs were placed in a patient with Sjogren syndrome for treatment of keratoconjunctivitis sicca. More than 12 years later, the plug was noted to have eroded through the canaliculus and extruded through the conjunctiva and lid. RESULTS: The extruded plug was removed, and punctal cautery was performed on 2 other puncta with improved symptom control. CONCLUSIONS: Plug extrusion through the conjunctiva and lid is a possible consequence of punctal plug placement.

Chronic parotitis with multiple calcifications: Clinical and sialendoscopic findings.

OBJECTIVES/HYPOTHESIS: To characterize clinical, imaging, and sialendoscopy findings in patients with chronic parotitis and multiple parotid calcifications. STUDY DESIGN: Retrospective review. METHODS: Clinical history, radiographic images and reports, lab tests, and operative reports were reviewed for adult patients with chronic parotitis and multiple parotid calcifications who underwent parotid sialendoscopy. RESULTS: Thirteen of 133 (10%) patients undergoing parotid sialendoscopy for chronic sialadenitis had more than one calcification in the region of the parotid gland. Seven patients (54%) were diagnosed with immune-mediated disease from autoimmune parotitis (positive Sjögren's antibodies or antinuclear antibodies) or human immunodeficiency virus (HIV) disease. The six patients (46%) who did not have an immune-mediated disorder had most calcifications located anterior or along the masseter muscle. Eight of 13 patients (61%) had at least one calculus found in the parotid duct on sialendoscopy. Four patients (38%) had multiple punctate calcifications within the parotid gland, all of whom had either autoimmune parotitis or HIV. None of the proximal or punctate parotid calcifications posterior to the masseter were visualized on sialendoscopy. CONCLUSIONS: Chronic parotitis in conjunction with multiple parotid calcifications is uncommon and was identified in 10% of our cohort. We contrast two classifications of parotid calcifications: 1) intraductal stones that cause recurrent duct obstruction and are often located within the main parotid duct along or anterior to the masseter and 2) punctate intraparenchymal parotid gland calcifications that are not visualized on sialendoscopy and may represent underlying inflammatory disease. LEVEL OF EVIDENCE: 4 Laryngoscope, 127:1565-1570, 2017.

Rumpel-Leede Phenomenon in a Patient with Laboratory Markers Positive for Sjögren Disease.

Rumpel-Leede phenomenon is a rare clinical sign involving the appearance of purpura after application of a tourniquet or in noninvasive arterial blood pressure monitoring. This sign has been most commonly associated with hypertensive and diabetic microvascular fragility and thrombocytopenia. We describe a case of Rumpel-Leede phenomenon in an otherwise healthy patient with positive laboratory markers for Sjögren disease, a previously undescribed relationship. We aim to inform physicians of this potential complication in patients with Sjögren disease and suggest special consideration be given to patients with autoimmune diseases with secondary vascular or dermal manifestations.

Dental Implants in Patients with Sjögren's Syndrome.

BACKGROUND: Limited evidence is available for applying dental implants in Sjögren's syndrome (SS) patients. PURPOSE: This study aims to retrospectively assess clinical outcome of implant therapy in a cohort of well-classified patients with SS. MATERIALS AND METHODS: All SS patients attending the University Medical Center Groningen for follow-up (n = 406) were asked whether they had implants. In SS patients with implants peri-implant health and implant survival was recorded and compared with data from matched healthy controls. Patients' symptoms, health-related quality of life, oral functioning, and satisfaction were assessed using validated questionnaires. RESULTS: Of the responding SS patients (n = 335), 21% was provided with implants. Of these 69 SS patients, 50 SS patients were willing to join our study. In SS patients, peri-implant health was reasonably good with minor marginal bone loss and a peri-implantitis prevalence of 14%, comparable with healthy controls. Implant survival was 97% (median follow-up 46 months) [IQR 26;73], and overall patients' satisfaction was high. Oral functioning correlated negatively with dryness, patients' satisfaction, and chewing ability in SS patients. CONCLUSIONS: Implant therapy is common in our cohort of SS patients. In spite of shortcomings of our retrospective analysis, implants in SS patients seem to perform comparable with implants in healthy patients.

Dental implants in patients with oral mucosal diseases - a systematic review.

To reveal dental implants survival rates in patients with oral mucosal diseases: oral lichen planus (OLP), Sjögren's syndrome (SjS), epidermolysis bullosa (EB) and systemic sclerosis (SSc). A systematic literature search using PubMed/Medline and Embase databases, utilising MeSH and search term combinations identified publications on clinical use implant-prosthetic rehabilitation in patients with OLP, SjS, EB, SSc reporting on study design, number, gender and age of patients, follow-up period exceeding 12 months, implant survival rate, published in English between 1980 and May 2015. After a mean observation period (mOP) of 53·9 months (standard deviation [SD] ±18·3), 191 implants in 57 patients with OLP showed a survival rate (SR) of 95·3% (SD ±21·2). For 17 patients with SjS (121 implants, mOP 48·6 ± 28·7 months), 28 patients with EB (165 implants, mOP 38·3 ± 16·9 months) and five patients with SSc (38 implants, mOP 38·3 ± 16·9 months), the respective SR was 91·7 ± 5·97% (SjS), 98·5 ± 2·7% (EB) and 97·4 ± 4·8% (SSc). Heterogeneity of data structure and quality of reporting outcomes did not allow for further comparative data analysis. For implant-prosthetic rehabilitation of patients suffering from OLP, SjS, EB and SSc, no evidence-based treatment guidelines are presently available. However, no strict contraindication for the placement of implants seems to be justified in patients with OLP, SjS, EB nor SSc. Implant survival rates are comparable to those of patients without oral mucosal diseases. Treatment guidelines as for dental implantation in patients with healthy oral mucosa should be followed.

The Association Between Sjögren Syndrome and Adverse Postoperative Outcomes: A Historical Cohort Study Using Administrative Health Data.

BACKGROUND: Sjögren syndrome is a chronic autoimmune disorder of the exocrine glands associated with cardiovascular events. We aimed to evaluate postoperative complications in patients with Sjögren syndrome undergoing noncardiac surgery. Specifically, we tested the primary hypothesis that patients with Sjögren syndrome have a greater risk of postoperative cardiovascular complications than those without the disease. Our secondary hypotheses were that patients with Sjögren syndrome are at greater risk of thromboembolic complications, microcirculatory complications, and mortality. METHODS: We obtained censuses of 2009 to 2010 inpatient hospital discharges across 7 states. Sjögren syndrome was identified by the present-on-admission diagnosis code 710.2. Each Sjögren n syndrome discharge was propensity matched to 4 control discharges. A generalized linear model was used to compare matched Sjögren syndrome patients and controls on risk of in-hospital cardiovascular complications, thromboembolic complications, microcirculatory complications, and mortality. RESULTS: Among 5.5 million qualifying discharges, our final matched sample contained 22,785 matched discharges, including 4557 with Sjögren syndrome. Sixty-six (1.45%) of the matched discharges with Sjögren syndrome and 213 (1.17%) of the matched controls had associated in-hospital cardiovascular complications. The adjusted odds ratio (99% confidence interval) was estimated at 1.14 (0.79-1.64), which was not statistically significant (P = 0.35). There were no significant differences in the odds of in-hospital thromboembolic complications (1.12 [0.82-1.53]; P = 0.36), in the odds of in-hospital microcirculatory complications (0.98 [0.77-1.26]; P = 0.86), or in the odds of in-hospital mortality (1.11 [0.76-1.61]; P = 0.49). CONCLUSIONS: The presence of Sjögren syndrome does not place patients at an increased risk for postoperative complications or in-hospital mortality.

Effect of Lid Debridement-Scaling in Sjögren Syndrome Dry Eye.

PURPOSE: To evaluate the effect of lid debridement-scaling (LDS) on dry eye signs and symptoms in subjects with Sjögren syndrome (SS). METHODS: This prospective randomized controlled study enrolled 14 female subjects with SS. Seven subjects were randomized into the treatment group where they were selected to receive LDS; the remainder did not receive LDS and served as control subjects. Lid debridement-scaling was conducted using a stainless steel golf club spud (Hilco Wilson Ophthalmics, Plainville, MA) on both the upper and lower eyelids of both eyes. Outcome variables were assessed before LDS and again 1 month later. The outcome variables were the Ocular Surface Disease Index (OSDI), Symptom Assessment iN Dry Eye (SANDE) visual analog scores, ocular staining (SICCA OSS [Sjögren's International Collaborative Clinical Alliance Ocular Staining Score]), fluorescein tear breakup time (FLBUT), meibomian gland score (MGS), meibomian gland yielding liquid secretions (MGYLS) score, and line of Marx's (LOM) position. RESULTS: Thirteen subjects completed the study. Data from only the right eye were analyzed. For the control group (n = 6; mean [± SD] age, 62.3 [± 11.6] years), the pre-LDS, post-LDS, and significance level (pre-LDS mean [± SD] vs. post-LDS mean [± SD]; p value) were as follows: OSDI (58.3 [± 22.1] vs. 48.3 [± 29.0]; p = 0.051), SANDE (77.4 [± 22.1] vs. 89.6 [± 32.6]; p = 0.20), SICCA OSS (7.0 [± 4.5] vs. 8.2 [± 3.5]; p = 0.25), MGS (1.3 [± 1.5] vs. 1.0 [± 0.9]; p = 0.75), MGYLS (0.3 [± 0.5] vs. 0.0 [± 0.0]; p = 0.50), FLBUT (2.99 [± 1.54] vs. 2.85 [± 1.79]; p = 0.63), and LOM (2.0 [± 0.0] vs. 2.0 [± 0.0]; p = n/a). For the treatment group (n = 7; mean [± SD] age, 58.0 [± 8.1] years), the pre-LDS, post-LDS, and significance level were as follows: OSDI (63.2 [± 13.3] vs. 46.9 [± 19.4]; p = 0.04), SANDE (72.6 [± 17.1] vs. 77.0 [± 28.0]; p = 0.54), SICCA OSS (6.6 [± 2.9] vs. 5.0 [± 3.9]; p = 0.02), MGS (1.0 [± 1.2] vs. 3.1 [± 1.7]; p = 0.01), MGYLS (0.0 [± 0.0] vs. 0.6 [± 1.0]; p = 0.50), FLBUT (3.13 [± 0.81] vs. 3.45 [± 1.03]; p = 0.53), and LOM (0.9 [± 0.9] vs. 1.0 [± 1.0]; p = 1.00). CONCLUSIONS: This pilot study showed that LDS improved symptoms, ocular staining, and meibomian gland function for the group that received LDS. This indicates that LDS can aid in the management of SS dry eye.

A case of marginal zone B cell lymphoma mimicking IgG4-related dacryoadenitis and sialoadenitis.

BACKGROUND: IgG4-related dacryoadenitis and sialoadenitis (IgG4-DS), so-called Mikulicz's disease, is characterized by elevated serum IgG4 and infiltration of IgG4-positive plasma cells in glandular tissues. Recently, several studies reported both malignant lymphoma developed on the background of IgG4-associated conditions and IgG4-producing malignant lymphoma (non-IgG4-related disease). CASE PRESENTATION: We report on the case of a 70-year-old man who was strongly suspected IgG4-DS because of high serum IgG4 concentration (215 mg/dl) and bilateral swelling of parotid and submandibular glands. Biopsies of cervical lymph node and a portion of submandibular gland were performed. These histopathological findings subsequently confirmed a diagnosis of marginal zone B cell lymphoma. CONCLUSION: Differential diagnosis of IgG4-DS is necessary from other disorders, including Sjögren's syndrome, sarcoidosis, Castleman's disease, Wegener's granulomatosis, lymphoma, and cancer. We suggest that biopsy of swollen lesions is important for a definitive diagnosis of IgG4-DS and discuss the mechanism of development in this case.

Kidney biopsy findings in primary Sjögren syndrome.

BACKGROUND: Renal involvement is rare in primary Sjögren syndrome (PSS). In this study, we examined renal biopsy findings in patients with PSS and correlated them with their clinical and renal findings. METHODS: Twenty-five patients with PSS who underwent renal biopsies from two renal units in Scotland between 1978 and 2013 were identified from renal biopsy database. We examined the renal morphologic, clinical and renal findings at the time of renal biopsy, renal and patient outcomes. RESULTS: The diagnosis of PSS preceded renal biopsy in 18/25 patients. In this group, the median duration of the disease was 5.5 years. Significant proteinuria, combined microscopic haematuria and proteinuria and reduced renal excretory function were found in 76, 56 and 84% of patients, respectively. The 3-year actuarial patient survival was significantly lower in patients with glomerulonephritis as compared with tubulointerstitial nephritis (66 versus 100%, P = 0.02). There was no difference in 3-year actuarial renal survival between these two groups (92 versus 92%, P = 1.0). CONCLUSIONS: Renal biopsy is rare in PSS and often reveals diverse pathological findings. Glomerulonephritis, as compared with tubulointerstitial nephritis, is associated with higher early mortality. Further studies are needed to evaluate the utility of renal biopsy and its impact on disease management.

The role of parotidectomy in Sjögren's syndrome.

Sjögren's syndrome, a chronic and progressive autoimmune disorder mainly characterized by xerophthalmia, xerostomia, and parotid enlargement, is primarily managed medically, but some patients will require surgical management. Patients with Sjögren's syndrome have an increased risk of non-Hodgkin lymphoma. Superficial parotidectomy is indicated for diagnostic purposes and can be therapeutic in limited circumstances. Surgical indications for parotidectomy in Sjögren's syndrome include recurrent parotitis refractory to medical management; salivary gland malignancy; and severe, refractory pain. Surgical complications include transient or permanent facial nerve injury, post-operative pain, persistent inflammation of remnant parotid tissue, Frey syndrome, and facial scarring.

Chronic inflammation in the pancreas and salivary glands--lessons from similarities and differences in pathophysiology and treatment modalities.

The pancreas and salivary glands have similar anatomical structures and physiological functions producing bicarbonate-rich fluid containing digestive enzymes and other components to be delivered into the gut. Despite these similarities, the two organs are also different in numerous respects, especially regarding the inflammatory diseases affecting them. This article will summarize the pathophysiology and current and potential pharmacological treatments of chronic inflammatory diseases such as chronic pancreatitis, autoimmune pancreatitis, Sjögren's syndrome and irradiation-induced salivary gland atrophy. Despite the differences, in both organs the inflammatory process is accompanied by epithelial tissue destruction and fibrosis. Both in pancreatic and in salivary research, an important task is to stop or even reverse this process. The utilization of stem/progenitor cell populations previously identified in these organs and the application of mesenchymal stem cells are very promising for such regenerative purposes. In addition, gene therapy and tissue engineering research progressively advance and have already yielded clinically beneficial preliminary results for salivary gland diseases. For the hard-to-access, hard-to-regenerate pancreas these developments may also offer new solutions, especially since salivary and pancreatic progenitors are very similar in characteristics and may be mutually useful to regenerate the respective other organ as well. These novel developments could be of great significance and may bring new hope for patients since currently used therapeutic protocols in salivary and in pancreatic chronic inflammatory diseases offer primarily symptomatic treatments and limited beneficial outcome.

[MALT-type parotid lymphoma--a case report and the review of the literature]. / Chloniak typu MALT slinianki przyusznej--opis przypadku i przeglad pismiennictwa.

Primary lymphomas of the salivary glands are rare. It is estimated that they constitute no more than 5% of all lymphomas in different locations. The most common subtype developing in parotid glands is marginal zone B-cell mucosa associated lymphoid tissue type lymphoma (MALT) that belongs to a group of low-grade tumours. There are many factors associated with the incidence of that proliferative process: environmental and infectious agents as well as immune deficiency states. We describe a case of primary non-Hodgkin's lymphoma of the parotid gland arising in the background of previously undiagnosed and untreated Sjögren's syndrome in a 52-year-old woman. The article concerns a short review of the literature regarding etiology, symptoms, treatment and survival prognosis in that rare disease as well. MALT lymphomas should always be considered in the differential diagnosis of the tumors and swelling of the parotid gland area. A special, regular monitoring should include all patients with Sjögren's syndrome as those with the proven greater risk of developing that proliferative disease. The role of the laryngologist in the case of MALT-type lymphoma of the parotid gland should focus on a diagnosis and possible tumor cytoreduction with maximal saving of the facial nerve. The essential treatment of this pathology is one of the oncologists and haematologists.