Association of Preterm Birth with Inflammatory Bowel Disease and Salivary Gland Disease: Machine Learning Analysis Using National Health Insurance Data.

This study employs machine learning and population data for testing the associations of preterm birth with inflammatory bowel disease (IBD), salivary gland disease, socioeconomic status and medication history, including proton pump inhibitors. The source of population-based retrospective cohort data was the Korea National Health Insurance Service claims data for all women aged 25-40 years and who experience their first childbirths as singleton pregnancy during 2015 to 2017 (402,092 women). These participants were divided into the Ulcerative Colitis (UC) Group (1782 women), the Crohn Group (1954 women) and the Non-IBD Group (398,219 women). For each group, the dependent variable was preterm birth during 2015-2017, and 51 independent variables were included. Random forest variable importance was employed for investigating the main factors of preterm birth and testing its associations with salivary gland disease, socioeconomic status and medication history for each group. The proportion of preterm birth was higher for the UC Group and the Non-IBD Group than for the Crohn Group: 7.86%, 7.17% vs. 6.76%. Based on random forest variable importance, salivary gland disease was a top 10 determinant for the prediction of preterm birth for the UC Group, but this was not the case for the Crohn Group or the Non-IBD Group. The top 5 variables of preterm birth for the UC Group during 2015-2017 were socioeconomic status (8.58), age (8.00), proton pump inhibitors (2.35), progesterone (2.13) and salivary gland disease in 2014 (1.72). In conclusion, preterm birth has strong associations with ulcerative colitis, salivary gland disease, socioeconomic status and medication history including proton pump inhibitors.

Inner ear salivary gland choristoma extending to the middle ear with congenital profound hearing loss and facial palsy: a case report.

BACKGROUND: Salivary gland choristoma (SGCh) is a rare benign tumor reported in several unusual sites, such as the gastrointestinal tract, the optic nerve, and the internal auditory canal, but never reported in the inner ear. CASE PRESENTATION: An 8-year-old girl with a history of left profound congenital hearing loss presented to us with ipsilateral progressive severe facial nerve palsy (House-Brackmann Grade VI). The left tympanic membrane was swollen with a pulsatile tumor. Radiological investigations revealed a multilocular tumor in the inner ear extending into the middle ear and internal auditory canal (IAC). We performed a partial resection of the tumor by transmastoid approach to preserve the anatomical structure of the facial nerve. The tumor was pathologically diagnosed as SGCh. Two years after surgery, her facial function recovered to House-Brackmann Grade II and the residual tumor did not show regrowth on MRI. CONCLUSIONS: Although the natural course of this rare tumor is unknown, a partial resection is an acceptable treatment procedure when functional recovery of the facial nerve is anticipated.

[Raynaud phenomenon with arterial thromboses and IgG4-related disease]. / Phénomène de Raynaud avec thromboses artérielles au cours d'une maladie associée aux IgG4.

INTRODUCTION: This is a clinical case illustrating a diagnosis of an IgG4 related-disease (IgG4-RD) diagnosed in a vascular context. CASE REPORT: A 47-year-old man with no past medical history consulted for a recent and disabling Raynaud phenomenon without trophic disorder. Vascular examinations revealed multiple arterial thromboses with no abnormal finger and toe pressures. Secondly, weight loss and submandibular glands enlargement appeared, leading to the diagnosis of IgG4-RD without a link being able to be established with vascular involvement. This is the second observation of this association. A French translation of the new classification criteria for IgG4-RD published in 2019 by the American College of Rheumatology and European Ligue Against Rhumatism (ACR/EULAR) is offered with direct application to the clinical case. CONCLUSION: A Raynaud phenomenon with distal arterial thrombosis is rarely observed in the IgG4-RD.

Patients with non-Sjögren's sicca report poorer general and oral health-related quality of life than patients with Sjögren's syndrome: a cross-sectional study.

Understanding the impact of the disease on quality of life is crucial in patient management. In this cross-sectional study, general and oral health-related quality of life questionnaires, and thorough examinations of oral and ocular dryness were performed in age- and sex-matched patients with primary Sjögren's syndrome (pSS group), non-Sjögren's syndrome sicca (non-SS group) and healthy controls. General and oral health-related quality of life were investigated with the 36-Item Short Form Health Survey and the 14-Item Oral Health Impact Profile questionnaires, respectively. Subjective symptoms of xerostomia and ocular dryness were recorded using the Summated Xerostomia Inventory and Ocular Surface Disease Index, respectively. Clinical examinations included evaluation of clinical oral dryness scores, candida counts, unstimulated and stimulated saliva secretory rates, tear osmolarity, tear film break-up time, Schirmer I test and ocular surface staining. Both patient groups had pronounced signs and symptoms of xerostomia and ocular dryness. Even though the non-SS patients had less severe clinical signs than the pSS patients, they demonstrated much poorer general and oral health-related quality of life. In conclusion, non-SS patients require more attention in order to improve their quality of life.

Salivary gland performance in autoimmune diseases: review and meta-analysis.

Objectives: Autoimmune diseases include a spectrum of disorders in which immune response to the autoantigens leads to tissue damage or dysfunction. Xerostomia, salivary gland dysfunction and lack of saliva are some common symptoms associated with many autoimmune diseases.Methods: In this review study, the meta-analysis technique is used to objectively review the relationship between autoimmune diseases and salivary gland dysfunction. We have searched Medline and Embase and Google Scholar. By Revman 5.3, meta-analysis was performed to investigate the salivary flow rate in both stimulatory and non-stimulatory saliva. The sample size obtained from these studies was 130 people with autoimmune diseases and 100 healthy individuals.Results: The results showed a significant decrease in the level of non-stimulatory saliva in people with autoimmune diseases.Conclusions: A complete and comprehensive understanding of the clinical manifestation of systemic diseases is crucial in early diagnosis of diseases and identifying the mechanisms that develop the disease. Other than xerostomia, there is a significant reduction in salivary flow rate in patients with autoimmune diseases. As saliva plays a very important role in oral health and has significant functions, more attention is needed for monitoring and managing of hyposalivation in autoimmune patients.

Presentation is key to diagnosing salivary gland disorders.

Initial signs and symptoms offer the best guide to next steps in assessment, testing, and treatment, plus any needed referral or multidisciplinary care.

Multimodal ultrasound investigation (grey scale, Doppler and 2D-SWE) of salivary and lacrimal glands in healthy people and patients with diabetes mellitus and/or obesity, with or without sialosis.

AIM: To evaluate the ultrasound (US) modifications [grey scale, Doppler, 2D-share wave elastography (2D-SWE)] ofsalivary (parotid and submandibular) and lacrimal glands in healthy people and patients with diabetes mellitus and/or obesity, with or without sialosis. MATERIAL AND METHODS: We evaluated 170 patients (1020 glands, 1700 grey scale and Doppler images), split in two groups (group 1- healthy people, group 2- obese and/or diabetes patients, with or without sialosis). For each patient we assessed the parotid, submandibular and lacrimal glands in grey scale US (echogenicity, homogeneity, glandular contour, posterior border, lymph nodes), color Doppler US and 2D-SWE. All images were analyzed by two examiners blinded to each other or to patients. RESULTS: The interobserver agreement was strong or moderate for all parameters. In group 2, the salivary glands had increased echogenicity, homogeneous aspect and invisible posterior border (all p<0.001). There was no significant variation of elasticity modulus in the groups analyzed (5.46±1.57 vs 5.67±1.81 in parotid, 8.63±1.84 vs 8.55±1.94 in submandibular and 9.47±2.1 vs 9.53±2.23 in lacrimal glands, all p>0.05) or according to the body mass index (BMI), sex, patient age, the aspect in grey scale/Doppler US or the presence of sialosis (all p>0.05). CONCLUSION: The main US differences between healthy people and patients with diabetes mellitus and/or obesity are suggested by the echogenicity, homogeneity, posterior border and the size of glandular area. No significant differences of elasticity modulus were found between the analyzed groups or related to BMI, sex, patient age or other grey scale/Doppler US items analyzed.

Ectopic Germinal Center-Like Structures in Minor Salivary Gland Biopsy Tissue Predict Lymphoma Occurrence in Patients With Primary Sjögren's Syndrome.

OBJECTIVE: To determine risk factors for primary Sjögren's syndrome (SS)-associated lymphoma in a multicenter cohort of patients, with analysis of the predictive power of previously reported risk factors, including the presence of ectopic germinal center (GC)-like structures in minor salivary gland (MSG) biopsy tissue. METHODS: One hundred fifteen patients with primary SS were included, and MSG biopsy tissue from these patients was retrospectively examined, focusing on the presence of ectopic GC-like structures. Epidemiologic, clinical, biologic, immunologic, and histologic data were collected at the time of diagnosis of primary SS. Patients with non-Hodgkin's lymphoma (NHL) were compared with those without NHL during the follow-up period, using a Cox proportional hazards multiple regression model. RESULTS: NHL was diagnosed in 8 patients (6.96%), and ectopic GC-like structures in 19 patients (16.5%). The presence of ectopic GC-like structures was associated with a 7.8-fold increased risk of lymphoma occurrence (95% confidence interval [95% CI] 1.73-34.86 [P = 0.0075]). Other independent predictors included a positive cryoglobulin test result (hazard ratio [HR] 7.10, 95% CI 1.74-28.92 [P = 0.006]), male sex (HR 28.73, 95% CI 4.46-144.87 [P = 0.0004]), sensorimotor neuropathy (HR 35.48, 95% CI 5.79-217.39 [P = 0.0001]), and splenomegaly (HR 19.9, 95% CI 4.4-90 [P = 0.0001]). CONCLUSION: The presence of ectopic GC-like structures in MSG biopsy tissue is associated with the risk of lymphoma in patients with primary SS. These data reinforce the major role of MSG biopsy tissue in primary SS, for the identification a priori of a subgroup of patients with the highest risk of lymphoma.

High-throughput RNA sequencing reveals distinct gene signatures in active IgG4-related disease.

We aimed to characterize the molecular differences and effects from prednisone treatment among IgG4-related disease with salivary gland lesions (RD-SG), without SG lesions (RD-nonSG), and IgG4-related retroperitoneal fibrosis (RF). RNA sequencing was conducted on blood from 25 RD-SG, 11 RD-nonSG, 3 RF and 10 control subjects. Among these, 8 RD-nonSG and 12 RD-SG patients were subjected to treatment with prednisone and/or glucocorticoid-sparing agents. Six RD patients had a longitudinal time point. The mRNA levels of IgG4 and IgE, genes specific for Th2 cells, eosinophils, and neutrophils were over-expressed in RD-SG and RD-nonSG. A B-cell signature was suppressed in patients group versus controls, while Th1, Th2, Treg, and eosinophil gene signatures were increased in patients without treatment. Interestingly, Tfh genes and B cell signature were decreased at flare disease state. Prednisone treatment led to increased neutrophil, but decreased Treg signatures. Serum IgG4 levels correlated with the eosinophil and neutrophil gene signatures in RD-SG patients, and with a B cell signature in only RD-nonSG patients. IgG4, IgE, and cell-specific signatures are regulated in patients, suggesting the imbalance of immune and inflammatory cells in IgG4-related disease. Prednisone treatment selectively modulates Treg, eosinophil, and neutrophil signatures.

The presence of minor salivary glands in the peritonsillar space.

Peritonsillar abscess (PTA) is traditionally considered only a purulent complication of acute tonsillitis (AT), but may be related to infection of minor salivary glands. We analysed the presence of peritonsillar minor salivary glands and inflammation patterns in 114 adult tonsils representing three patient groups: recurrent AT, chronic tonsillitis (CT), and PTA. Samples acquired from elective tonsillectomies were stored in formalin, and after preparation were microscopically examined for inflammation and fibrotic changes. Clinical features and histological characteristics were compared between the groups. Of all tonsils, the minor salivary glands were present in 77 (67.5%). Glands located near the tonsillar tissue showed signs of infection in 73 (94.8%), while only 3 (15.0%) of 20 glands located deeper in the peritonsillar space were infected. Compared to patients with recurrent AT and CT, those with PTA more often presented with periductal inflammation, p < 0.011 (PTA 82.1%, AT 42.9%, and CT 63.6%). The majority of our 114 tonsillectomy specimens, collected from patients with AT, CT, or PTA, presented with infected minor salivary glands, and inflammation of the peritonsillar space glands was evident. To further elucidate the association between these glands and PTA, tonsillar samples should be collected and analysed from patients during the acute phase of infection.

Simultaneous, congenital, mucocoele of the Blandin-Nühn glands and teratoma of the tongue: early management and follow up at one year.

Mucocoele of the Blandin-Nühn glands and teratomas of the tongue are rare in newborn babies. We present what is to our knowledge the first documented case of both at the same time, and describe management and follow up over 12 months after the birth. Prenatal diagnosis defined the treatment plan during delivery to establish an airway for the baby promptly. A normal delivery is possible if the cyst is anterior, and can be punctured during delivery. Delayed excision reduces the possibility of oedema and obstruction of the airway, ensures complete resection, avoids recurrence, and confirms the diagnosis. Mucocoeles are classified as extravasation or retention types; our observation supports the hypothesis of a "malformative type". A long follow-up is essential to monitor macroglossia, open bite, and the development of the mandible.

Diagnostic work-up in obstructive and inflammatory salivary gland disorders.

Inflammatory and obstructive disorders of the salivary glands are caused by very different pathological conditions affecting the gland tissue and/or the excretory system. The clinical setting is essential to address the appropriate diagnostic imaging work-up. According to history and physical examination, four main clinical scenarios can be recognised: (1) acute generalised swelling of major salivary glands; (2) acute swelling of a single major salivary gland; (3) chronic generalised swelling of major salivary glands, associated or not with "dry mouth"; (4) chronic or prolonged swelling of a single major salivary gland. The algorithm for imaging salivary glands depends on the scenario with which the patient presents to the clinician. Imaging is essential to confirm clinical diagnosis, define the extent of the disease and identify complications. Imaging techniques include ultrasound (US), computed tomography (CT) and magnetic resonance (MR) with MR sialography.

Eosinophilic sialodochitis: redefinition of 'allergic parotitis' and 'sialodochitis fibrinosa'.

Sialodochitis fibrinosa and allergic parotitis have described rare patients with recurrent salivary gland swelling and mucus plugs, often with atopy. We have evaluated three patients with atopic disease, recurrent salivary gland swelling, and an eosinophilic sialodochitis. Two had eosinophil-rich mucus plugs. Fifty-six additional cases were identified in a medical literature database search, each defined by recurrent salivary gland swelling associated with eosinophil-rich mucus plugs or sialodochitis with periductal eosinophilic infiltration. The majority (78%) were reported from Japan. Females were predominantly affected (F:M = 2.3) with a median age of 47 years at evaluation. The parotid and submandibular glands were involved, respectively, in 71% and 46%. Allergic symptoms were present in 66%, atopic disease in 63% of those with reported allergy testing, and blood eosinophilia in 71%. Contrast sialography and other imaging modalities documented ductal dilatation in 82%. Treatments included anti-allergic medications (58%), systemic glucocorticoids (25%), duct cannulation with irrigation, steroid injection, and/or duct dilatation (36%), and glandular resection (19%). We recommend the diagnosis 'eosinophilic sialodochitis' be applied to patients who meet this case definition. The disease is a unique cause of chronic recurrent salivary gland swelling. Its likely allergic etiology may be amenable to current or future biologic therapies.

Peritonsillar Abscess: Complication of Acute Tonsillitis or Weber's Glands Infection?

OBJECTIVE: To review the literature concerning the 2 primary hypotheses put forth to explain the pathogenesis of peritonsillar abscess: "the acute tonsillitis hypothesis" (peritonsillar abscess is a complication of acute tonsillitis) and "the Weber gland hypothesis" (peritonsillar abscess is an infection of Weber's glands). DATA SOURCES: PubMed, EMBASE. REVIEW METHODS: Data supporting or negating one hypothesis or the other were elicited from the literature. CONCLUSIONS: Several findings support the acute tonsillitis hypothesis. First, the 2 main pathogens in peritonsillar abscess have been recovered from pus aspirates and bilateral tonsillar tissues with high concordance rates, suggesting that both tonsils are infected in patients with peritonsillar abscess. Second, studies report signs of acute tonsillitis in the days prior to and at the time of peritonsillar abscess. Third, antibiotic treatment reduces the risk of abscess development in patients with acute tonsillitis. However, some findings suggest involvement of the Weber's glands in peritonsillar abscess pathogenesis. First, high amylase levels have been found in peritonsillar pus. Second, the majority of peritonsillar abscesses are located at the superior tonsillar pole in proximity of the Weber's glands. We propose a unified hypothesis whereby bacteria initially infect the tonsillar mucosa and spread via the salivary duct system to the peritonsillar space, where an abscess is formed. IMPLICATIONS FOR PRACTICE: Our findings support the rationale for antibiotic treatment of patients with severe acute tonsillitis to reduce the risk of abscess development. Improved understanding of peritonsillar abscess pathogenesis is important for the development of efficient prevention strategies.

Are the patients with anatomic variation of the sublingual/Wharton's duct system predisposed to ranula formation?

OBJECTIVE: To evaluate ranula development according to anatomic variation of the ductal system of sublingual gland (SLG), especially the presence of Bartholin's duct. METHODS: The anatomic variation of SLG duct was prospectively investigated and compared between 55 consecutive patients with ranulas treated by SLG excision (group 1) and another 15 consecutive patients undergoing similar surgeries for other conditions (group 2). The ductal structures of SLGs and submandibular glands (SMG) were also compared between the pediatric patients and adult patients with ranulas. RESULTS: In 32 of 55 patients with ranulas (58.2%) and 1 of 15 patients without ranulas (6.7%), the SLG showed an anatomic variation of the main duct called Bartholin's duct structure (P<0.01). Seventeen of 22 (77.3%) pediatric patients with ranulas had Bartholin's ducts and 15 of 33 (45.5%) adult patients with ranulas had Bartholin's ducts (0.01

Relationships between oral health, dysphagia and undernutrition in hospitalised elderly patients.

OBJECTIVE: The aim of this study was to describe relationships between oral status, dysphagia and malnutrition in a hospitalised older people. BACKGROUND: Undernutrition in older people is a major concern in geriatric hospital wards. Different factors can modify nutritional status like dysphagia or oral status. MATERIALS AND METHODS: About 159 consecutive inpatients (108 women, 51 men) were examined. Comprehensive gerontological data at baseline and nutritional status according to BMI, MNA and serum albumin concentration, dependency according to ADL scores, dietary intake, swallowing capacities and oral status were collected. Swallowing capacities and dietary intake were reassessed 1 week after. RESULTS: Mean age was 85.28 (SD 5.68). Seventy-seven patients were malnourished (MNA) and 34 had dysphagia. Oral treatment was necessary in 142 patients (89.30% of all population). Candidiasis was present in 17 patients and salivary flow reduction in 50. Patients with dysphagia had the lowest dietary intake. After 1 week, patients with dysphagia were retested and dysphagia had abated in three of them. Dysphagia and undernutrition were associated (p < 0.001), and both were related to candidiasis (p < 0.001 and p < 0.01). Dysphagia was also related to salivary hypofunction (p < 0.001), loss of posterior occluding pairs (POPs; p = 0.014), oral self-care dependency (p < 0.001) and self-feeding dependency (p < 0.001). Salivary hypofunction was related to candidiasis (p < 0.001) and loss of POPs (p < 0.05), and candidiasis to loss of POPs (p < 0.01). CONCLUSION: Although no causality can be demonstrated, poor oral health was strongly associated with malnutrition, emphasising the importance to develop oral care strategies and to incorporate a dental examination into comprehensive gerontological assessment.

Rapid development of a simple ranula in a child.

OBJECTIVE: A simple ranula is a gradually progressive, benign condition of the salivary gland. The report here describes a very rare case of a simple ranula enlarging in an acute fashion, resulting in airway obstruction. CASE REPORT: A 7-year-old girl presented with swelling in the floor of the mouth, pushing the tongue upward and backward, requiring immediate nasotracheal intubation. Magnetic resonance imaging suggested a simple ranula requiring urgent surgery. The patient was followed up 4 weeks after discharge, at which time she was doing well. CONCLUSION: To the best of the authors' knowledge, this is the first case reported of a simple ranula presenting in an acute fashion, leading to airway compromise in a pediatric patient. The literature is reviewed and features of diagnosis and treatment are presented.