Rate of Submandibular Gland Atrophy Secondary to Chronic Sialolithiasis.

The rate of salivary gland atrophy secondary to chronic obstructive sialolithiasis has not been well-documented. The combination of 5 imaging studies over 12 years in a patient with repeat imaging for an unrelated pathology provides a unique opportunity to assess glandular atrophy over time. We hope that this case will support previous literature with an in vivo representation of the rate of glandular atrophy.

Glycyrrhizic acid ameliorates submandibular gland oxidative stress, autophagy and vascular dysfunction in rat model of type 1 diabetes.

The burden of diabetes mellitus (DM) and associated complications is increasing worldwide, affecting many organ functionalities including submandibular glands (SMG). The present study aims to investigate the potential ameliorative effect of glycyrrhizic acid (GA) on diabetes-induced SMG damage. Experimental evaluation of GA treatment was conducted on a rat model of type I diabetes. Animals were assigned to three groups; control, diabetic and GA treated diabetic groups. After 8 weeks, the SMG was processed for assessment of oxidative stress markers, autophagy related proteins; LC3, Beclin-1 and P62, vascular regulator ET-1, aquaporins (AQPs 1.4 and 5), SIRT1 protein expressions in addition to LC3 and AQP5 mRNA expressions. Also, parenchymal structures of the SMG were examined. GA alleviated the diabetes-induced SMG damage via restoring the SMG levels of oxidative stress markers and ET-1 almost near to the normal levels most probably via regulation of SIRT1, AQPs and accordingly LC-3, P62 and Beclin-1levels. GA could be a promising candidate for the treatment of diabetes-induced SMG damage via regulating oxidative stress, autophagy and angiogenesis.

Sialo-cutaneous fistula with ectopic submandibular gland sialolith, revealing a hidden ipsilateral enlarged and elongated styloid process: a consideration based on CT findings.

Sialolithiasis is a common salivary pathology, and an uncommon complication of sialadenitis and sialolithiasis is the formation of fistulous tracts to other compartments. Submandibular gland sialo-oral fistulae are not particularly remarkable, given the location of the gland and Wharton's duct, but submandibular sialolith-associated fistulae to other cervico-facial compartments (transcervical sialo-cutaneous and sialo-pharyngeal fistulae) are much less common. We report herein an unusual case of a 49-year-old obese man with sialo-cutaneous fistula containing a large, ectopic sialolith in subcutaneous tissue that was expected to undergo spontaneous elimination, but revealed hidden Eagle syndrome featuring an ipsilateral enlarged, elongated styloid process. Furthermore, we offer a thorough review of the literature regarding sialo-fistulae and highlight the relationship between an abnormal styloid process and submandibular sialadenitis with sialolithiasis and new tract formation based on computed tomography.

Giant torus mandibularis causing submandibular duct obstruction and sialadenitis.

Torus mandibularis is a benign osseous overgrowth arising from the lingual surface of the mandible. It is a common, incidental finding on imaging due to its relatively high prevalence. In the majority of cases, mandibular tori are asymptomatic. We report a novel presentation of a giant torus mandibularis causing bilateral obstruction of the submandibular ducts and consequent sialadenitis. Our patient presented with progressive pain centered in the floor of his mouth and had bilateral submandibular glandular enlargement on exam. Computed tomography showed a giant right torus mandibularis, which was causing obstruction and dilation of the bilateral submandibular ducts. Although conservative management was attempted, he ultimately underwent surgical resection of his torus with symptomatic improvement. This patient highlights a novel complication of torus mandibularis and illustrates successful treatment. Though not previously described, this complication may be underreported and should be considered in the appropriate clinical setting.

Sialographic Analysis of Radioiodine-Associated Chronic Sialadenitis.

OBJECTIVES/HYPOTHESIS: To apply a novel sialography classification system to identify parotid and submandibular ductal findings following I-131 therapy and to assess correlates to dose and duration of symptoms. STUDY DESIGN: Retrospective single-center case series. METHODS: Patients who underwent sialography between February 2008 and February 2019 after previously receiving I-131 treatment were identified via a retrospective chart review. Their sialograms were systematically evaluated and scored by applying the Iowa parotid sialogram scale to also include submandibular gland analysis. RESULTS: From 337 sialograms, 30 (five submandibular, 25 parotid) underwent analysis. Ductal stenosis was identified in all sialograms and was graded as moderate (>50%-75%) in 7/30 cases and severe (>75%) in 15/30 cases. The distal (main) duct was narrowed in 23/30 cases. No association was identified between degree of ductal stenosis and I-131 dose (P = .39), age (P = .81), or time from I-131 therapy to sialogram (P = .97). CONCLUSIONS: The Iowa parotid sialogram scale was successfully applied to report abnormalities of the parotid and submandibular ductal system. The most common manifestation of I-131-associated sialadenitis was a severe stenosis within the distal salivary duct. No statistically significant association was found between degree of ductal stenosis and dose of I-131, age, or duration of symptoms. LEVEL OF EVIDENCE: 4 Laryngoscope, 131:E1450-E1456, 2021.

Switching to normal diet reverses kwashiorkor-induced salivary impairments via increased nitric oxide level and expression of aquaporin 5 in the submandibular glands of male Wistar rats.

Kwashiorkor, a form of malnutrition, has been shown to cause impaired salivary secretion. However, there is dearth of information on the mechanism that underlies this complication. Also, whether returning to normal diet after kwashiorkor will reverse these complications or not is yet to be discerned. Thus, this study aimed at assessing the mechanisms that underlie kwashiorkor-induced salivary impairments and to evaluate the effects of switching back to normal-diet on kwashiorkor-induced salivary impairments. Weaning rats were randomly divided into 3 groups (control group, kwashiorkor group (KG), re-fed kwashiorkor group (RKG)) of 7 rats each. The control group had standard rat chow while the KG and RKG were fed 2% protein diet for 6 weeks to induce kwashiorkor. The RKG had their diet changed to standard rat-chow for another 6 weeks. Blood and stimulated saliva samples were collected for the analysis of total protein, electrolytes, amylase, immunoglobulin A (IgA) secretion rate, leptin, and ghrelin. Tissue total protein, nitric oxide level, expressions of Na+/K+-ATPase, muscarinic (M3) receptor, and aquaporin 5 in the submandibular glands were also determined. Data were presented as means ± SEM and compared using ANOVA with Tukey's post hoc test. RKG showed improved salivary function evidenced by reduced salivary lag-time and potassium and increased flow rate, sodium, amylase, IgA secretion rate, leptin, submandibular nitric oxide level, and aquaporin 5 expression compared with KG. This study for the first time demonstrated that kwashiorkor caused significant reduction in salivary secretion through reduction of nitric oxide level and aquaporin 5 expression in submandibular salivary glands. Normal-diet re-feeding after kwashiorkor returned salivary secretion to normal.

Sialendoscopy for Definitive Management of a Submandibular Abscess following Radiotherapy for Oropharyngeal Squamous Cell Carcinoma.

BACKGROUND/AIMS: Sialendoscopy has as yet been shown to be ideal for the management of sialolithiasis and chronic inflammatory diseases of the salivary gland. However, its applicability to the management of a broad range of salivary gland disease is continually growing. METHODS: Here we present a case report where sialendoscopy was used to successfully manage an intraparenchymal submandibular gland abscess in a patient with oropharyngeal squamous cell carcinoma managed with primary chemoradiation. RESULTS: The use of sialendoscopy enabled visualization of the patency of salivary ducts, drainage of abscess, and irrigation of antibiotic-impregnated fluid. In this particular patient, we were able to avoid a transcervical approach through a previously irradiated field, which would have necessitated concurrent tracheostomy and placed undue risk to surrounding neurovascular structures. CONCLUSION: Sialendoscopy should thus, in select patients, be considered as an initial intervention for patients with intraparenchymal salivary gland abscesses in which prior therapy creates an increased risk of complication from an open transcervical approach.

Submaxilitis aguda secundaria a la toma de azatioprina en paciente con enfermedad de Crohn / Azathioprine-induce acute submandibular sialadenitis in a patient with Crohn's disease

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Mumps presenting with unilateral, synchronous parotid and submandibular gland swelling.

A previously healthy 22-year-old woman presented with acute, unilateral facial and neck swelling, associated with fever and malaise. She was initially treated with intravenous antibiotics; however, CT imaging showed unilateral, synchronous swelling and inflammation of the parotid and submandibular glands, and a PCR swab from the parotid duct was positive for mumps. She was fully immunised and had no contact in the preceding period with anyone diagnosed with mumps. She responded to supportive management and her symptoms resolved over the course of her admission. Unilateral, synchronous swelling and severe inflammation of both the parotid and submandibular glands in mumps is a very unusual presentation, and not one previously reported in the literature.

Sialendoscopic removal of fish bone-induced sialoliths in the duct of the submandibular gland.

Obstructive sialadenitis of the submandibular gland is commonly caused by sialoliths, but more rarely by foreign body-induced sialoliths. Here, we report minimally invasive sialendoscopic removal of fish bone-induced sialoliths in the duct of the submandibular gland. A 43-year-old woman presented with recurrent swelling of the right submandibular gland at other hospital. Computed tomography (CT) showed an 8-mm linear calcification in the posterior part of Wharton's duct. The lesion was deemed difficult to remove and she was followed up. However, because the lesion did not resolve spontaneously within 9 months and chronic sialadenitis symptoms persisted, she was referred to our department for endoscopic removal. CT showed a linear calcification (5.6×1.2×0.8mm) connecting 2 spherical calcifications (2.3×2.1×1.9mm; 1.8×1.4×1.1mm) in the anterior part of Wharton's duct. The patient underwent endoscopic removal of the lesion using a 1.6-mm-diameter sialendoscope under local anesthesia. The specimen contained a fish bone connecting 2 sialoliths. The patient was unaware of the fish-bone injury. After removal, there was no recurrence of submandibular gland swelling during 6 months follow-up.

Acute submaxillitis: Etiological data and etiological management.

OBJECTIVES: The objective of the present study was to test the hypothesis that acute submaxillitis involves salivary duct obstruction. Obstacle characteristics and treatment were analyzed. MATERIAL AND METHODS: Patients presenting with an episode of acute submaxillitis between 2009 and 2015 were retrospectively included. All underwent salivary duct imaging and/or sialendoscopy, with pathologic examination if the salivary gland was removed for etiologic diagnosis. For etiological treatment, if the causal lesion could not be treated by isolated sialendoscopy, surgery used a direct approach assisted by sialendoscope. In case of failure of these procedures, submaxillectomy was indicated. RESULTS: Twenty-nine patients were included, 28 of whom showed salivary duct abnormality. At least 1 calculus was found in 27 patients; calculi were usually single (n=20), situated in the mid-third (n=21), and large (mean 7.7mm). Ten patients showed stenosis, associated with salivary calculus in 9 cases. Twenty-five patients with salivary duct obstruction underwent sialendoscopy. Isolated sialendoscopy was used in 5 cases, and a combined approach in 13 cases. CONCLUSION: An episode of submaxillitis requires salivary duct exploration by sialendoscopy, to enable early treatment given the prevalence of associated calculi and high success rate of conservative management by sialendoscopy.

Chronic sialadenitis due to the stone inside the accessory duct of submandibular gland.

PURPOSE: Sialolithiasis is the most common cause of chronic sialadenitis. In this case report, intraoperative finding of an accessory submandibular duct, obstructed with stone, originating from the same gland nearby the main Warthon's duct, is presented. CASE REPORT: A 22-year-old male patient, suffering from eating-related pain and swelling in his left submandibular region, was diagnosed with left sublandibular gland sialadenitis with radiologically manifested sialolithiasis, and gland excision was advised. Surgery was performed under general anesthesia. When the full anatomical scenery was delineated before excision of the gland, we surprisingly encountered two submandibular ducts originating from ipsilateral gland, one of them was obstructed with stone. After two ducts were ligated, the gland with sialolith was excised. According to histopathologic examination, the duct obstructed with stone was identified as the accessory duct and the other one was the main Wharton's duct. Postoperative days were uneventful; no neurologic complication was observed. CONCLUSIONS: Otolaryngologists should be aware of anatomic variations of the submandibular duct(s) to avoid possible complications, especially intraoperatively, because rutine preoperative radiologic preparation does not include investigation of possible accessory ducts.

Cystatin S-a candidate biomarker for severity of submandibular gland involvement in Sjögren's syndrome.

Objectives: Salivary cystatin S is a defence protein mainly produced by submandibular glands and involved in innate oral immunity. This study aimed to verify whether cystatin S was diversely expressed in different disease subsets of primary Sjogren's syndrome (pSS) patients, defined on the basis of salivary flow [unstimulated salivary flow rate (USFR)], minor salivary gland (MSG) focus score and submandibular gland ultrasonography abnormalities. We also evaluated miR-126 and miR-335-5p expression in MSG biopsies to verify whether an aberrant regulation of cystatin S at the glandular level may influence its salivary expression. Methods: Forty pSS patients and 20 sex- and age-matched healthy volunteers were included. Salivary cystatin S levels were assessed by western blot analysis using a stain-free technology. The expression of miR-126, miR-335-5p and cystatin S was assessed by quantitative PCR in 15 MSG biopsies differing for USFR and MSG focus score. Results: We found that salivary cystatin S was significantly decreased in pSS patients vs healthy volunteers ( P = 0.000), especially in those with hyposalivation. A positive correlation was observed between cystatin S and USFR ( r = 0.75, P = 0.01). Salivary cystatin S was also significantly reduced in patients with a submandibular gland ultrasonography score ⩾2. The expression levels of miR-126 and miR-335-5P increased in inverse proportion with USFR. The mRNA of cystatin S did not change significantly, suggesting post-transcriptional regulation. Conclusion: Cystatin S emerged as a promising biomarker for pSS, strongly correlated with glandular dysfunction. An upregulation of miR-126 and miR-335-5P might be implicated in its expression.

A case of acute postoperative transitory sialadenosis of the submandibular glands in a healthy dog.

A 1-year-old healthy female cross-breed dog, weighing 4.5 kg, was scheduled for elective neutering. Fentanyl (5 µg/kg) and propofol (4 mg/kg) were administered intravenously (IV) to induce anesthesia, which was maintained with isoflurane and a constant fentanyl infusion rate (10 µg/kg/hr). During the recovery from the anesthesia, the presence of bilateral dense submandibular masses was recognized, as was the excessive secretion of saliva. An ultrasound examination was performed and revealed bilateral abnormally-diffused enlargement of the submandibular salivary glands. A cytology examination was conducted, and no signs of abnormality were found. The size of the swellings subsequently diminished, completely subsiding after 2 hr, as did the hyper-salivation. To the authors' knowledge, this represents the first case report of an acute transient swelling of submandibular glands after general anesthesia in a dog.

Association of serum electrolytes and smoking with salivary gland stone formation.

To further define potential factors that may contribute to stone formation in salivary glands (sialolithiasis), a retrospective chart review was performed of patients diagnosed with sialolithiasis between March 1, 1998 and February 29, 2012. Information on salivary gland stone number, location and size, medical history, medications, and serum electrolyte levels were collected. Associations between electrolyte levels and stone characteristics (such as stone number and size) were examined. Fifty-nine patients were identified; their median age was 58 years (range 25-89 years) and most were male (95%). Salivary stones were most commonly located in the submandibular glands (83%). Thirty-five patients (59%) had a smoking history, with 16 (27%) reported as current smokers. There was a significant association between current smoker status and stone size (mean largest stone size 12.4±8.8mm vs. 7.5±4.8mm in current smokers vs. non-smokers; P=0.03). Serum sodium levels (r=0.32, P=0.014) and serum potassium levels (r=0.31, P=0.017) showed significant positive correlations with stone size. While the aetiology of sialolithiasis remains unclear, smoking (which can contribute to reduced saliva flow) and higher serum sodium levels (which can reflect volume depletion) are associated with larger salivary stones.

Prevalence of Submandibular Gland Synucleinopathy in Parkinson's Disease, Dementia with Lewy Bodies and other Lewy Body Disorders.

BACKGROUND: Clinical misdiagnosis, particularly at early disease stages, is a roadblock to finding new therapies for Lewy body disorders. Biopsy of a peripheral site might provide improved diagnostic accuracy. Previously, we reported, from both autopsy and needle biopsy, a high prevalence of submandibular gland synucleinopathy in Parkinson's disease (PD). Here, we report on an extension of these studies to subjects with dementia with Lewy bodies (DLB) and other Lewy body disorders in 228 autopsied subjects from the Arizona Study of Aging and Neurodegenerative Disorders. OBJECTIVE: To provide an estimate of the prevalence of histological synucleinopathy in the submandibular glands of subjects with PD and other Lewy body disorders. METHODS: Submandibular gland sections from autopsied subjects were stained with an immunohistochemical method for α-synuclein phosphorylated at serine 129. Included were 146 cases with CNS Lewy-type synucleinopathy (LTS), composed of 46 PD, 28 DLB, 14 incidental Lewy body disease (ILBD), 33 Alzheimer's disease with Lewy bodies (ADLB) and 2 with progressive supranuclear palsy and Lewy bodies (PSPLB). Control subjects included 79 normal elderly, 15 AD, 12 PSP, 2 conticobasal degeneration (CBD) and 2 multiple system atrophy (MSA). RESULTS: Submandibular gland LTS was found in 42/47 (89%) of the PD subjects, 20/28 (71%) DLB, 4/33 (12%) ADLB and 1/9 (11%) ILBD subjects but none of the 110 control subjects. CONCLUSIONS: These results provide support for further clinical trials of in vivo submandibular gland diagnostic biopsy for PD and DLB. An accurate peripheral biopsy diagnosis would assist subject selection for clinical trials and could also be used to verify other biomarkers.