Nuances and Management of Hilar Submandibular Sialoliths With Combined Transoral Robotic Surgery-Assisted Sialolithotomy and Sialendoscopy.

OBJECTIVE: To describe the management, technical nuances, and success rates of transoral robotic surgery (TORS)-assisted sialolithotomy. STUDY DESIGN: Retrospective database review. SETTING: Quaternary academic medical center. METHODS: Between the months of January 2015 and May 2019, patients with hilar submandibular gland stones underwent 2 main variations of TORS-assisted sialolithotomy and sialendoscopy: (1) TORS followed by sialendoscopy for patients with palpable predominantly single stones and (2) either sialendoscopy followed by TORS and sialendoscopy or sialendoscopy followed by TORS only for patients with nonpalpable or multiple stones. Clinical charts were reviewed to collect data, including stone size (imaging review, intraoperative measurement), palpability, duration of operation, TORS variation, operative challenges, symptom improvement, gland preservation rate, and complications. RESULTS: Thirty-seven patients were identified. Patients were 26 to 80 years old (mean, 57.2 years), and 40.5% were female. Twenty-four patients (64.9%) underwent TORS followed by sialendoscopy; 10 (27.0%), sialendoscopy followed by TORS and sialendoscopy; and 3 (8.1%), sialendoscopy followed by TORS only. The mean stone size was 12.4 mm (range, 4-28 mm). Eleven patients had multiple stones with a mean 4 stones per patient (range, 2-9). Procedural success was 91.9% (34/37) at a mean follow-up of 34.2 weeks (range, 1.4-262.1), and the gland preservation rate was 97.3% (36/37). No patients reported symptoms of lingual nerve injury at 3-month follow-up. CONCLUSION: TORS combined with sialendoscopy for hilar submandibular gland sialolithiasis allows for improved visualization of critical anatomy, tissue manipulation, and operative flexibility. In our experience, the operative success rate is high, and duration of surgery compares favorably with conventional combined hilar approaches.

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 plus laser lithotripsy in sialolithiasis of the submandibular gland in 64 patients: A simple and safe procedure.

OBJECTIVE: To demonstrate the safety and efficiency of holmium laser-assisted lithotripsy during sialendoscopy of the submandibular gland using a retrospective, interventional consecutive case series. METHODS: We performed 374 sialendoscopies between 2008 and 2015 and evaluated all patients regarding clinical symptoms, clinical findings, therapy and outcome. We performed 109 procedures of holmium laser-assisted lithotripsy in 64 patients whose sialoliths measured 5 mm or more in diameter. In addition to retrospective case note reviews, we performed telephone interviews of all patients in January 2017. RESULTS: We performed 374 consecutive submandibular gland sialendoscopy procedures in 276 patients between 2008 to 2015. Sialolithiasis had either previously been diagnosed, or symptoms highly suggestive of sialolithiasis of the submandibular gland presented in 197 patients. Holmium laser-assisted Laser lithotripsy was performed in 109 cases (64.9%). Smaller mobile concrement was removed directly either by forceps or wire basket, or following marsupialisation of the submandibular duct. This was the case in 88 patients (29.1%). Three patients (0.8%) required surgical removal of the submandibular gland due to early abscess. The majority of patients (n = 374 procedures; 90.1%) remained symptom-free after two or more years following intervention. In the remaining procedures (n = 37 procedures; 9.9%), patients reported discreet postprandial problems but did not seek medical attention. In total, we managed to preserve the submandibular gland and avoid open surgery in 99% of patients through endoscopic management of submandibular concrement and duct stenosis. CONCLUSION: Holmium laser-assisted lithotripsy is a simple, safe, and effective procedure for treating patients with sialolithiasis of the submandibular gland. Removal of the gland is rarely required, and removing the gland without prior sialendoscopy is no longer recommended. It should be offered to all patients with submandibular gland sialolithiasis, or such patients should be referred to the appropriate centre for sialendoscopy before submandibulectomy is considered.

Accessory submandibular duct sialolithiasis identified during sialendoscopy.

We describe a unique case of a 62-year-old patient with recurrent right submandibular sialadenitis. He initially appeared to have extensive sialolithiasis of the right submandibular duct on computed tomography imaging and examination, but sialendoscopy demonstrated a normal-appearing right submandibular duct. An accessory duct posterior and parallel to the normal duct was identified at the time of sialendoscopy and was found to have extensive sialolithiasis, which required removal by both an endoscopic and intraoral technique. To the best of our knowledge, this is the first report of sialolithiasis of an accessory submandibular duct identified at the time of sialendoscopy; the other few reported cases in the literature are based primarily on findings from traditional sialography or magnetic resonance sialography. Identification of accessory salivary ducts requires intraoperative consideration of this entity during sialendoscopy. Open approaches to sialolithiasis, however, may be aided by appropriate preoperative imaging.

Nodular oncocytic hyperplasia: Can cytomorphology allow for the preoperative diagnosis of a nonneoplastic salivary disease?

BACKGROUND: Nodular oncocytic hyperplasia (oncocytosis) of the salivary glands is a benign process that does not inherently require surgical excision. However, cytologic findings in fine-needle aspiration (FNA) of oncocytosis cases have not been well characterized previously, limiting preoperative identification. METHODS: All available cases of oncocytosis with corresponding FNA specimens were identified from the pathology archives of 3 academic institutions. Clinical, cytologic, and histologic findings were tabulated for all cases. RESULTS: Twelve cases of oncocytosis were identified from 11 patients, including 11 parotid FNA specimens and 1 submandibular FNA specimen. On the original diagnoses, 6 specimens were classified as benign, 4 as atypical, and 2 as nondiagnostic. Oncocytosis was listed in the differential diagnosis in only 1 case. Among diagnostic aspirates, 8 demonstrated low cellularity and 2 demonstrated moderate cellularity. All 10 cases demonstrated oncocytic cells in small to medium groups, with single cells in just 1 case. Spindled and squamous morphology were each noted in 3 cases. Four cases demonstrated cystic change and 1 showed background mucin without goblet cells. No necrosis or mitoses were observed. CONCLUSIONS: Although oncocytosis demonstrates some overlap with Warthin tumor and oncocytoma, it lacks the diagnostic findings specific to oncocytic salivary gland malignancies such as salivary duct carcinoma, acinic cell carcinoma, mammary analog secretory carcinoma, and mucoepidermoid carcinoma. Despite current limitations in the understanding of oncocytic salivary gland lesions, the presence of a paucicellular specimen comprised of small groups of oncocytic cells should raise the possibility of oncocytosis in the differential diagnosis and can favor it in elderly patients with multiple salivary nodules. Cancer Cytopathol 2017;125:627-34. © 2017 American Cancer Society.

Bilateral subacute lacrimal gland enlargement mimicking dacryoadenitis in a 7-year-old boy: a rare adverse effect of valproic acid (sodium valproate).

A healthy 7-year-old boy presented with bilateral symmetrical lacrimal gland enlargement; a week later salivary gland enlargement was also noted. Clinical investigations suggested no diagnosis, and surgical biopsy was considered. Valproic acid (sodium valproate), which he was taking for absence seizures, has been reported to cause salivary gland swelling in adults. Suspecting that a similar mechanism could be causal, the drug was discontinued. Complete resolution of the lacrimal and salivary gland enlargement rapidly ensued. This is the first report of lacrimal gland enlargement caused by valproic acid.

Sialoadenitis por sialolito inusual de la glándula submaxilar derecha en un adulto / Sialadenitis due to uncommon sialolith of the right submaxillary gland in an adult patient

Se presenta el caso clínico de un paciente de 45 años de edad, atendido en el Departamento de Cirugía Maxilofacial del Hospital Provincial Docente Clinicoquirúrgico Saturnino Lora Torres por presentar molestias en el suelo de la boca con 3 años de evolución. Después de un estudio minucioso se comprobó que se trataba de una sialoadenitis por sialolitiasis de la glándula submaxilar derecha. Se realizó sialolectomía canalicular intrabucal. El cálculo extraído tenía forma cónica, más ancho en su porción posterior, de 3,8 cm de largo y 4,2 cm de circunferencia externa, color amarillo-verdoso y consistencia dura friable

The case report of a 45 years patient was presented. He was assisted in the Maxillofacial Surgery Department of Saturnino Lora Torres Clinical Surgical Provincial Hospital in Santiago de Cuba due to discomfort in the floor of the mouth with a course of 3 years. After a detailed study it was confirmed that it was a sialadenitis due to sialolithiasis of the right submaxillary gland. An intraoral canalicular sialolectomy was carried out. The stone removed had a conic form, wider in its posterior portion, 3.8 cm long and 4.2 cm in the external circumference, yellow-green color and friable hard consistency

Idiopathic Sialolithiasis: Scalpel Versus Current Trends in Management.

Idiopathic sialolithiasis is one of the most common affliction of the salivary glands with the submandibular gland most frequently involved. The article discusses the occurrence and incidence of this condition with a review of different diagnostic and treatment modalities. In light of several advances to diagnose and treat sialolithiasis, a simple sialolithotomy for idiopathic submandibular gland sialolithiasis may prove to be a preferred treatment for selected patients as it remains a cost-effective and simple out-patient department procedure with minimal/no complications and a highly satisfactory outcome. For stones slightly proximal in the Wharton duct, a "dual" approach of milking the gland to bring the stone more distally followed by a sialolithotomy with a scalpel proves to be a quick and effective procedure.

Evaluation of Sialendoscopy-Assisted Treatment of Submandibular Gland Stones.

PURPOSE: The aim of the present study was to evaluate the therapeutic efficiency of sialendoscopy-assisted operations in the treatment of submandibular gland stones. MATERIALS AND METHODS: The data from 8 patients with sialolithiasis who underwent sialendoscopy from August 2015 to January 2016 at the Department of Oral and Maxillofacial Surgery, School of Stomatology, China Medical University (Shenyang, China) were retrospectively reviewed. All the patients had undergone preoperative technetium-99m pertechnetate salivary gland scintigraphy. The results revealed that the salivary glands exhibited normal or slightly reduced uptake and excretion dysfunction. Computed tomography examinations revealed stones located in the intraductal area near the glands or in the branches that could not be removed owing to their deep locations within the mouth. Therefore, an endoscope was inserted, the stones were located intraductally using sialendoscopy, and a transcervical incision was made to remove the stones and preserve the submandibular gland. RESULTS: The stones were completely removed, and the submandibular gland was preserved in all cases. The patients recovered well postoperatively, and no complications developed. CONCLUSIONS: Our results suggest that sialendoscopy-assisted sialolithectomy is an effective and safe surgical technique for the removal of proximal and intraglandular submandibular gland stones. The patients' quality of life had obviously improved postoperatively.

Rare Submandibular Presentation of Pediatric Castleman Disease: Case Report.

Castleman disease (CD), a benign lymphoproliferative disorder that affects lymph nodes, is uncommon in children, with only approximately 100 cases published. Although 23% of pediatric CD cases are found in the neck, there is no substantial reported percentage found in the salivary glands, especially the submandibular salivary gland (SMSG). A pediatric case of CD involving the SMSG is reported because of its extreme rarity.

Differential diagnosis of IgG4-related sialadenitis, primary Sjögren syndrome, and chronic obstructive submandibular sialadenitis.

Our aim was to differentiate IgG4-related sialadenitis, primary Sjögren syndrome, and chronic obstructive submandibular sialadenitis by analysing clinical, radiographic, and pathological features. Fifty-five patients, 50, and 50 were enrolled, respectively and their baseline characteristics and serological, sialographic, and pathological findings compared. The male:female ratio for IgG4-related sialadenitis was 1:1.2 for primary Sjögren syndrome 1:15.7, and for chronic obstructive submandibular sialadenitis1:0.92. Numbers with enlarged salivary glands were 55, 16, and 50; with xerostomia 26, 48, and 0; with a history of allergy 26, 4, and 6, and with coexisting systemic disease 12, 19, and 0 (p=0.14). Mean (SD) serum IgG4 concentrations were 109.1 (97.9), 4.9. (1.9) g/L, and 5.3 (1.6) g/L, p<0.001 in all cases. Sialography showed enlargement of the gland, dilatation of the duct, and slightly decreased secretory function in IgG4-related disease; obvious sialectasia and decreased secretory function in Sjögren syndrome; and dilatation of Wharton's duct and filling defects in obstructive sialadenitis. Histopathological examination showed lymphoplasmacytic infiltration with storiform fibrosis, lymphoplasmacytic inflammation and lymphoepithelial lesions, and dilatation of the duct with epithelial metaplasia in the three groups, respectively. The number of IgG4-positive plasma cells was 123 (45)/HPF, 8 (3)/HPF, and 5 (4)/HPF, while the IgG4-/IgG-positive cell ratio was 71.7 (13.9)%, 4.6 (2.5)%, 18.9 (19.7)%, respectively (p<0.001). The three conditions have different clinical, radiographic, and pathological features that provide important clues to the differential diagnosis. Serological and histological tests are important, and comprehensive consideration is necessary.

A Rare Case of Lateral Ectopic Thyroid.

Ectopic thyroid tissue is an abnormality caused by aberrant thyroid gland embryogenesis during its passage from the floor of the primitive foregut to its final position. The most frequent place of ectopic thyroid tissue is the base of tongue, whereas lateral thyroid gland is a very rare finding. The present case describes a case of thyroid dysgenesis, caused by a submandibular ectopic thyroid gland. Thyroid scintigraphy was crucial for the diagnosis: in fact, the patient was asymptomatic, and the diagnosis was performed only on the basis of subclinical hypothyroidism.

Sialodochitis fibrinosa (kussmaul disease) report of 3 cases and literature review.

BACKGROUND: Sialodochitis fibrinosa is a rare disease which is characterized by recurring episodes of pain and swelling of the salivary glands due to the formation of mucofibrinous plugs. Analytic studies ascertain elevated levels of eosinophils and immunoglobulin E (IgE). Imaging studies such as magnetic resonance imaging (MRI) and sialography reveal dilation of the main salivary duct (duct ectasia). Treatment is initially supportive, consisting of compressive massages, and use of antihistamines and/or corticosteroids. MATERIAL AND METHODS: In the following, 3 cases of sialodochitis fibrinosa are presented which were diagnosed in a third level hospital during the period of 2008 and 2016, as well as a literature review of all cases reported to our knowledge. RESULTS: Of the 41 cases found, including the 3 of this article, 66% were women with an average age of 45 years old. However, 75% of reported cases were of Japanese heritage. Involvement of the parotid glands was more frequent than the submandibular glands. In more than half of all cases treatment with compressive massages, antihistamines and/or corticosteroids was effective. CONCLUSION: Clinicians should consider sialodochitis fibrinosa as a diagnostic possibility when presented with cases of recurring parotid and submandibular gland tumescence.

Possible Drug-Associated Sialolithiasis From the Bicarbonate Anhydrase Inhibitor Topiramate: A Case Report and Literature Review.

Topiramate is an antiepileptic drug indicated for the treatment of seizure disorders, migraine prophylaxis, and, more recently, weight loss. This new indication will likely increase the use of this agent significantly. As a carbonic anhydrase inhibitor, topiramate can affect the pH of bodily fluids and is known to increase the risk of nephrolithiasis. However, as discussed in the present report, these properties also result in an as yet unaddressed risk of the development of sialoliths, calcified stones formed in the salivary duct or glands. The physiologic mechanisms for stone development in the salivary gland are reviewed and the pharmacologic effects of topiramate on sialolith formation discussed. The present report describes a female patient treated with topiramate for migraine prophylaxis who subsequently presented with a sialolith in the left submandibular duct.

Küttner Tumor: IgG4-Related Disease of the Submandibular Gland.

Küttner tumor/chronic sclerosing sialadenitis is a fibroinflammatory process that characteristically involves the submandibular gland of patients with IgG4-related disease. Histologic examination is often important to make the diagnosis because of its nonspecific clinical and radiologic findings. Microscopically, Küttner tumor should be distinguished from other entities such as extranodal marginal zone lymphoma, Sjögren's syndrome, and lymphoepithelial sialadenitis. The lesion is histologically well-demarcated with lobular architecture, extensive fibrosis, marked lymphoplasmacytic inflammation, formation of lymphoid follicles, acinar atrophy, and obliterative phlebitis, without the presence of lymphoepithelial lesions. The IgG4-to-IgG positive plasma cell ratio of >40 % is also an important feature to support the diagnosis of Küttner tumor. Moreover, flow cytometry is helpful to exclude a lymphoproliferative process. Clinicians and pathologists should consider the diagnosis of Küttner tumor in patients with elevated serum IgG4 level. Timely and accurate diagnosis is important for appropriate management.