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.

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.

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.

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.

Sialendoscopy-assisted treatment for radioiodine-induced sialadenitis.

PURPOSE: Chronic sialadenitis is a common complication of radioactive iodine for the treatment of thyroid disease. The aim of this study was to describe the authors' experience with interventional sialendoscopy for the management of radioiodine-induced sialadenitis. MATERIALS AND METHODS: Twelve patients with radioiodine-induced sialadenitis treated with sialendoscopy from January 2013 through December 2013 at the Department of Oral and Maxillofacial Surgery, School of Stomatology, China Medical University were retrospectively reviewed. Age, gender, and time to development of radioiodine-induced sialadenitis were obtained from the hospital database. All patients were asked to undergo visual analog scale (VAS) and salivary gland scintigraphy (SGS) examinations before and 6 months after surgery. A paired t test was conducted, and a P value less than .05 was considered statistically significant. RESULTS: Twelve patients (15 parotid glands and 4 submandibular glands) successfully underwent interventional sialendoscopy under local anesthesia. Ductal stenosis was the most common feature identified by endoscopy. Among the 12 patients, swelling occurred in 91.7%. Compared with the preoperative score of 6, the mean VAS score 6 months after sialendoscopy was 3; 15 glands (78.9%) showed improved uptake and excretion by SGS. The postoperative VAS score was significantly lower than the preoperative VAS score (P < .05), and the postoperative SGS result was significantly higher than the preoperative SGS result (P < .05). CONCLUSIONS: Interventional sialendoscopy could be an effective technique for the treatment of sialadenitis caused by radioactive iodine.

Frequency and outcomes of submandibular gland obstruction following resection of squamous cell carcinomas in the vicinity of the submandibular duct.

INTRODUCTION: The aim of this study was to report rates, associated factors and outcomes of submandibular gland obstruction following surgery for squamous cell carcinoma in the anterior floor of the mouth (FOM) or ventral tongue without a neck dissection where resection has involved (or has been in very close proximity to) the submandibular duct. METHODS: A retrospective case note review was carried out for the period January 2007 to December 2011. Only patients who received primary local surgical resection for squamous cell carcinoma of the anterior FOM or ventral tongue were included. RESULTS: Fifty-two patients were analysed. Fifteen (29%) developed submandibular gland symptoms following surgery. Symptoms resolved spontaneously for 14 (93%) and 1 patient required the submandibular duct to be repositioned. Comparatively, 2 patients (13%) developed symptoms when the duct was repositioned during primary surgery. No patients had their submandibular gland removed. CONCLUSIONS: Resection of small tumours associated with the FOM and ventral tongue in proximity to the submandibular duct is associated with obstructive symptoms in about a quarter of patients; this is reduced by half when redirecting the duct. Symptoms are self-limiting and self-resolving. A prospective clinical trial comparing duct repositioning with subcapsular gland excision would help clarify potential benefits and best treatment modalities.

Foreign body induced sialolithiasis treated by sialoendoscopic intervention.

OBJECTIVE: The study aims to identify the impact of sialolith formation by reviewing the foreign body induced sialolithiasis treated by sialoendoscopic intervention. METHODS: The study group included 13 patients whose sialolithiasis was induced by foreign body. After the routine radiographic examination, sialoendoscopic procedures were performed. Then, the treatment protocol was designed. RESULTS: The occupations of the 13 patients included 5 fishermen, 3 office workers, 2 workers, 1 teacher, 1 farmer, and 1 retired police officer. All patients had a unique diet habit-seafood. Eleven patients had a remembered incident of implanted fish bone and the following symptoms, with either obstructions or infections. Only 2 of the 13 had no memory of such an injury. All the stones were in the ducts of submandibular glands. In 10 procedures, there was 1 solitary stone, whereas 2 stones were encountered in 3 procedures. After being removed, 16 stones were crushed to expose the fish bone nidus of the stone. There was relief of symptoms after the procedures. CONCLUSIONS: This study supported the possibility that some sialoliths resulted from a retrograde migration within the salivary ducts. In our study, the occupations (fisherman), the diet habit (seafood), and the injury history (a remembered incident of implanted fish bone and the following symptoms) were obviously related to the stone formation that was induced by the fish bone.

Sialoendoscopy: a viable treatment for I(131) induced sialoadenitis.

To evaluate the viability, efficacy, and safety of sialoendoscopy for the diagnosis and management of radioiodine I(131-) related sialoadenitis, we retrospectively reviewed 30 patients referred between September 2007 and July 2013 from the Thyroid Surgery Unit to the Maxillofacial Unit of the Second University of Naples Hospital with persistent sialoadenitis after treatment with I(131). After the affected gland had been isolated, the endoscope was introduced into the duct under local anaesthesia with 2% lignocaine and continuous lavage with isotonic saline, and was advanced until it reached the ductal system. We studied 24 women and 6 men, mean (SD) age 52 (??) years. In 25 patients I(131) was given for papillary (83%), in 3 for medullary (10%), and in 2 for follicular thyroid carcinoma (7%). Stenosis alone was found in 30 glands (40%), mucous plugs alone in 35 (47%), and mucous plugs, stenosis, and kinks in 10 (13%). Of the 75 glands, dilatation of the ducts was successful in 70, and we completely removed all mucous plugs and kinks. We achieved symptomatic improvement in 23 patients (77%) during a follow-up ranging from 2 weeks to 84 months. Sialoendoscopy is a viable technique for the diagnosis of obstructive salivary disease, and is a safe and effective way to treat sialoadenitis, the most common complication of treatment with I(131).

[Swelling of the neck following tonsillectomy. Lateral cervical fistula]. / Zervikale Schwellung nach Tonsillektomie. Laterale Halsfistel.

A 16-year-old patient presented with recurrent cervical swelling to the right side of the neck on coughing and sneezing. Although present since childhood, the symptoms had progressed over the preceding year. Immediately prior to this period a bilateral tonsillectomy had been performed for recurrent tonsillitis. Magnetic resonance imaging revealed a complete lateral cervical fistula extending between the thyroid and submandibular glands on the right side of the neck. Successful surgical resection accomplished complete removal of the fistula.

[The new S2k AWMF guideline for the treatment of obstructive sialadenitis in commented short form]. / Die neue S2k AWMF Leitlinie zur Behandlung der obstruktiven Sialadenitis in kommentierter Kurzform.

A new and interdisciplinary S2k AWMF guideline for the treatment of obstructive sialadenitis has been published. There have been several technical achievements, for instance in the field of ultrasonography, via sialendoscopy, or by MR-sialography, that have increased the possibilities for diagnosis and treatment of patients with obstructive sialadenitis. In the past, the treatment of choice in case of unsuccessful medical treatment was a complete extirpation of the affected salivary gland. Nowadays, using a variety of modern treatment options (like sialendoscopy, or extracorporeal shock-waves lithotripsy sometimes combined with salivary duct incision), it is possible in most patients, especially in cases of sialolithiasis, to preserve the affected gland. A functional recovery after gland-sparing surgery is described but more data is needed to finally evaluate the long-time results. The new guideline describes all relevant steps to diagnose an obstructive sialadenitis and values all diagnostic tools critically. Finally, all recommendable therapy options are described and valued, too.

Submandibular hydatid cyst fistulized into the oral cavity.

UNLABELLED: Hydatid cysts of the head and neck are rare, even in regions where echinococcal infestation is endemic. Although complications, like cyst rupture and infection, may occur, an external fistula is extremely rare. This study examined a case of hydatid cyst that had fistulized in the right submandibular region of the oral cavity, which eroded the mandible. CASE REPORT: A 45 year-old female patient visited our clinic with complaints of a discharge that left a bad taste in her mouth that persisted for 1 month and a painless swelling in the right submandibular region that had slowly expanded over the past 5 months. A physical examination revealed a cystic mass located in the right submandibular region that had fistulized in the oral cavity. The results were consistent with a hydatid cyst. We performed submandibular gland exeresis and a marginal mandibulectomy to excise the hydatid cyst together with the fistulous tract.

ARFI quantitative elastography of the submandibular glands. Normal measurements and the diagnosis value of the method in radiation submaxillitis.

OBJECTIVES: Evaluation of Acoustic Radiation Force Impulse Imaging (ARFI) elastography performance in predicting the elasticity of the submandibular glands in normal situations and after radiation therapy. MATERIAL AND METHOD: A number of 54 normal submandibular glands from 27 voluntary subjects and 33 pathological submandibular glands (radiation submaxillities) from 18 patients who had undergone radiation therapy for various cervical and facial oncological conditions were included in study. All the patients had undergone a B mode ultrasonography (Tissue Harmonic Imaging, 8-14 MHz) while the submandibular volume was determined and subsequently an ARFI examination while the shear wave velocity (SWV) was measured (in the central, peripheral and subcapsular areas, with the results expressed in m/s). RESULTS: In the volunteers' group the mean value of the SWV of the left submandibular gland was 1.68 ± 0.46 m/s, determined in the centre of the gland, 1.88 ± 0.4 m/s in the periphery (corresponding to the subcapsular parenchyma) and the SWV of the right submandibular gland was 1.74 ± 0.35 m/s (centrally) and 1.84 ± 0.43 m/s in the periphery. The mean value of all measurements was 1.82 ± 0.41 m/s. The mean volume of the glands was 7.97 ± 2.63 cm3. In the group of patients who had underwent radiation therapy (at least 35Gy), the mean value of the SWV was 2.24 ± 0.49 m/s centrally and 2.1 ± 0.58 m/s in the periphery on the left and 1.99 ± 0.5 m/s centrally and 2.21 ± 0.52 m/s in the periphery on the right. The mean value of all the measurements was 2.13 ± 0.52 m/s and the mean volume of the gland was 5.95 ± 4.16 cm3. CONCLUSIONS: Elastography using ARFI technique is a valid examination in the evaluation of the normal and pathological submandibular gland stiffness. The values of the shear wave velocities that correspond to a normal stiffness, determined through the ARFI technique, are similar in the two glands. After cervical and facial radiation therapy the values of the SWV are increased, indicating a change in the consistency of the gland thus implying a structural transformation. The ARFI technique can be used in the evaluation of the salivary glands pathology.

A retrospective case series illustrating a possible association between a widened hilum and sialolith formation in the submandibular gland.

OBJECTIVE: The purpose of this study was to explore any association between anatomical variances in the ductal system and sialolith formation using sialoendoscopy and acrylic resin replication of the ductal system. METHODS: A retrospective study of 372 submandibular gland sialoendoscopies was performed to review the findings of the submandibular gland duct anatomy. Using sialoendoscopy and replicated casts, a high rate of hilar widening was noted in patients with submandibular sialolithiasis. RESULTS: Sialolithiasis was detected in 326 of the patients who presented with obstructive symptoms. Around 67% (285/426) of the stones were located in the distal third of the ducts or at the hilum of the submandibular gland. During the sialoendoscopic procedure, the anatomy of the ductal system was examined and 285/326 (87.4%) of the hilums were noted to be widened like a basin. The anatomy of the duct from the replicated casts demonstrated a treelike structure and the basin-like widening of the hilum was found in all the excised submandibular glands. CONCLUSION: Using sialoendoscopy, a high number of patients presenting with sialolithiasis in the submandibular gland seem to have an anatomical variance in the hilar region. The reproduced ductal system from excised glands also demonstrated this abnormal widening of the hilum. Although further studies need to be performed, we try and explain why there is such a high prevalence of hilar widening in patients with submandibular sialolithiasis.

The role of salivary duct morphology in the aetiology of sialadenitis: statistical analysis of sialographic features.

Morphologic characteristics of salivary ducts may contribute to stagnation of saliva. The authors hypothesized that some features might contribute to development of submandibular and parotid sialadenitis. 106 digital subtraction sialograms (DSS) were retrospectively reviewed for: degree of sialadenitis, length of Wharton's and Stensen's ducts (SD), and angle of Wharton's duct (WD) genu. Student's t test was used for independent samples to statistically compare normal and sialadenitis groups. The effect of independent variables (age, gender, side, degree of sialadenitis) on the dependent variable (length or angle) were tested using regression analysis. Submandibular duct sialadenitis was mild (67%), moderate (8%), or severe (25%); parotid duct sialadenitis was mild (57%), moderate (18%), or severe (25%). Mean length of normal WD was 58.2 mm, and 56.3 mm with sialadenitis. Mean length of normal SD was 52 mm, and 53 mm with sialadenitis. The mean angle of WD genu was 115° in normal ducts, and 119° with sialadenitis. None of the independent variables affected variation in length or angle. There were no statistical differences in duct length or measured angle between normal and sialadenitis groups. There is a wide variation in salivary duct morphology but this does not appear to be associated with the cause of sialadenitis.