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.

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.

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.

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.

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.

[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.

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.

Unusual presentations of disseminated histoplasmosis.

Histoplasmosis is considered to be the most prevalent endemic mycosis in United States that can present as a disseminated infection. The initial presentation of Disseminated Histoplasmosis (DH) can be atypical. We report three cases with such atypical presentation. Our first patient presented with bowel perforation, the second with left-sided pleural effusion and the third with submandibular abscess. Blood cultures as well as biopsy of perforation site, culture of pleural fluid and submandibular abscess were positive for Histoplasma Capsulatum (HC). We encourage clinicians to look for HC even in uncommon sites as dictated by the presenting symptoms and signs, especially in immunocompromised patients in endemic areas.

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.

Endoscopic observation and strategic management of obstructive submandibular sialadenitis.

PURPOSE: Obstructive submandibular sialadenitis is a relatively common disease in the clinic. The present study explored the cause and strategic management of chronic obstructive sialadenitis using sialoendoscopy and surgery. PATIENTS AND METHODS: From January 2005 to October 2007, 128 patients with obstructive symptoms in the submandibular gland were diagnosed using sialoendoscopy and occlusal films, and the obstructions were removed using interventional sialoendoscopy, surgery only, or combined techniques, depending on the size, shape, site, and quality of the sialolith in the duct. The shape and structure of the hilus of the submandibular gland was observed using sialoendoscopy. RESULTS: In the submandibular gland, the histologic features of 128 cases were identified endoscopically and radiographically. The endoscopic findings were of 3 types: sialolith in 114 (89%), mucus plug in 8 (6%), and stenosis in 6 (5%). Of 51 obstructions treated surgically, 47 were removed successfully, for a success rate of 92%. Of 63 obstructions treated using interventional sialoendoscopy, 52 were removed directly by sialoendoscopy, for a success rate of 83%. The unsuccessful cases were treated using sialoendoscopy or surgery. The obstructive symptoms were relieved in 12 of 14 patients without stones using dilation and irrigation under sialoendoscopy. Of the 114 patients with a stone, the sialoliths of 67 (59%) were located in the distal region (behind the first molar). The results showed that the more posterior the stone, the more difficult it was to remove. Three patients with treatment failure ultimately underwent resection of the gland. A basin-like structure in the hilus region was found in 86 (67%) of the 128 patients by sialoendoscopy. Finally, 5 patients were found to have a foreign body (fish bone) surrounded by a sialolith. CONCLUSIONS: Sialoendoscopy is a useful new technique, not to only detect the cause of obstruction in a submandibular gland, but also to treat the obstructive submandibular sialadenitis more effectively if incorporated with conventional surgical approaches.

Unilateral absence of submandibular gland secondary to stones. Aplasia versus early atrophy.

Major salivary gland absence is a rare disorder. The cause of congenital absence of the salivary glands has not been determined, but it may be associated with ectodermal defects of the first and second branchial arches. Isolated absence of a unilateral submandibular gland is an unusual entity with less than ten cases reported in the literature. The etiopathogenesis of isolated absence of a major salivary gland without other developmental anomalies is still unclear. The formation of a sialolith within the remaining Wharton's duct, associated with isolated aplasia (versus atrophy) of a unilateral submandibular gland has been recently reported. We describe in this work two cases of sialolithiasis within the ipsilateral remaining Wharton's duct in patients with isolated absence of a unilateral submandibular gland. In the cases reported, absence of the submandibular gland may have been the result of the complete acinar atrophy secondary to an early obstruction of Wharton's duct.

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.

Interventional sialoendoscopy: early clinical results.

PURPOSE: Sialoendoscopy is a novel minimally invasive technique to explore the salivary duct system and to treat obstructive salivary disease. This article describes the early clinical experience with endoscopic salivary duct exploration and sialolithectomy in 2 medical centers. PATIENTS AND METHODS: This is a retrospective case series of 94 patients, with submandibular (n = 77) or parotid (n = 17) sialadenitis secondary to sialolithiasis, strictures, or mucous plugs. Patients underwent sialoendoscopy at Baptist Hospital, Miami (n = 52) or at Massachusetts General Hospital, Boston (n = 42). Dilatation of the duct through the natural orifice was accomplished with salivary dilators. Three endoscope systems with diameters from 1.1 to 2.3 mm were used. Using a basket, grasper, lithotripsy, laser, or a combination of these, stones were fragmented or removed endoscopically. Strictures were dilated and mucous plugs removed. All cases were carried out under general anesthesia. RESULTS: Salivary duct navigation was accomplished in 91/94 patients. In 3 cases, duct dilatation was not possible due to scarring. Symptomatic relief was achieved in 81/91 patients (89.4%). Strictures and mucous plugs were visualized and managed in 18/18 patients. Sialoliths were visualized in 73 patients and stone fragmentation or retrieval was accomplished in 84.93% (62/73) of cases. Complications included 2 patients with temporary lingual nerve paresthesia and 1 patient with excess extravasation of irrigation fluid. CONCLUSION: The results of this study indicate that interventional sialoendoscopy is an effective, minimally invasive alternative treatment for obstructive salivary gland disease.