Transoral removal of submandibular hilar lithiasis: results on the salivary duct system, glandular parenchyma, and quality-of-life recovery.

OBJECTIVE(S): To confirm that hilar transoral submandibular sialolitectomy (TOSL) is the first treatment option for submandibular hilar lithiasis (SHL) in terms of glandular parenchyma recovery, salivary system restoration, and patient quality of life (QoL) improvement. METHODS: Depending on whether the stone was easily palpable, TOSL was carried out with or without sialendoscopy. For the first time in the literature, Magnetic Resonance Sialography (MR-Si) was performed before and after TOSL, to evaluate stone characteristics, glandular parenchyma status, hilum dilation and main duct recanalization. Radiological data was examined independently by two radiologists. COSQ, a recently validated and specific questionnaire, was used to assess associated QoL. RESULTS: Between 2017 and 2022, 29 TOSL patients were examined. With a high interobserver correlation, MR-Si was confirmed as a very useful radiological test in the pre- and post-surgical evaluation of SHL. The salivary main duct was completely recanalized in all cases. The presence of lithiasis was found in 4 patients (13.8%). After surgery, the majority of patients (79.31%) had hilum dilation. There was a statistically significant improvement in parenchyma status, but no significant progression to glandular atrophy. After surgery, COSQ mean values always improved (22.5 to 4.5). CONCLUSIONS: TOSL is the ideal surgical technique for the management of SHL, resulting in improved parenchymal inflammatory changes, recanalization of Wharton's duct, and enhancement patients' QoL. As a result, before removing the submandibular gland, TOSL should be considered as the first treatment option for SHL.

In Vivo Accuracy of Ultrasound for Sizing Salivary Ductal Calculi.

OBJECTIVES: The present study was developed to evaluate the accuracy of in vivo ultrasound sizing for parotid and submandibular salivary gland calculi, as compared with ex vivo pathology sizing with a standard plastic ruler after extraction. STUDY DESIGN: Retrospective chart review. SETTING: Ultrasound is frequently used to size salivary calculi and make treatment decisions, but the accuracy of measurements from this modality has not been validated. SUBJECTS AND METHODS: We evaluated and reviewed the charts and ultrasound examinations of 167 patients who underwent procedures for the treatment of sialolithiasis involving the parotid and submandibular glands. US examinations were performed between 2009 and 2016 in a tertiary-level hospital setting by the senior author. Measurements were collected from ultrasound evaluation before sialolithotomy, and pathology measurements were taken after removal. Ultrasound measurements in millimeters were compared with the measurements collected with a ruler. The differences were calculated and compared. RESULTS: A total of 167 calculi measurements were compared. Good concurrent validity between pathology and ultrasound measurements was suggested by a Pearson correlation of 0.92 (95% CI, 0.887-0.937). On Bland-Altman plot, correlation of the difference between US and pathology measurements showed a mean difference of 0.095 mm (95% CI, -0.19 to 0.38 mm) with a limit of agreement ranging from -3.59 mm (95% CI, -3.84 to -3.34 mm) to +3.78 mm (95% CI, +3.53 to +4.03 mm). CONCLUSIONS: Ultrasound is an accurate, relatively precise, and minimally invasive imaging tool for salivary gland sialolithiasis. Preoperative size of calculi can be used to guide management and clinical decision making. LEVEL OF EVIDENCE: 2C.

Ductal cells of minor salivary glands in Sjögren's syndrome express LINE-1 ORF2p and APOBEC3B.

BACKGROUND: Type I interferon activation is a hallmark event in Sjögren's syndrome. L1 retroelements stimulate plasmacytoid dendritic cells, activating the type I interferons, and are regulated by various mechanisms, including the APOBEC3 deaminases. As L1s are potential trigger factors in autoimmunity, we aimed to investigate the immunohistochemical localization of L1 ORF2p and its inhibitor APOBEC3B protein in minor salivary glands of Sjögren's syndrome patients. METHODS: Twenty minor salivary gland-tissue samples from 20 Sjögren's syndrome patients, classified according to Tarpley's histological criteria, and 10 controls were evaluated for L1 ORF2p and APOBEC3B expression via immunohistochemistry. RESULTS: L1 ORF2p was expressed in 17/20 SS patients and all controls. APOBEC3B expression was observed in 15/20 Sjögren's syndrome patients, 5/5 chronic sialadenitis, and 3/5 normal minor salivary glands. Both antibodies stained the cytoplasm of the ductal epithelial cells. Negative staining was observed in the acinar cells. L1 ORF2p-positive immunostaining was significantly lower in Tarpley IV Sjögren's syndrome patients than controls (P = .039), and APOBEC3B-positive staining was significantly lower in Tarpley I compared to Tarpley II Sjögren's syndrome patients (P = .008) and controls (P = .035). CONCLUSIONS: L1 ORF2p and APOBEC3B are expressed in the ductal epithelial cells of minor salivary glands that are among the key targets in Sjögren's syndrome. L1 ORF2p expression may promote the L1 ability to act as an intrinsic antigen in Sjögren's syndrome. The potential future use of L1 ORF2-reverse transcriptase inhibitors in autoimmunity supports further investigation of L1 epigenetic regulation by APOBEC3 enzymes.

[Giant sialolithiasis: Diagnosis and management. Case reports]. / Óriásnyálkövek: Diagnózis és kezelés.

Sialolithiasis is one of the most frequent form of calcifications in the maxillofacial area. 0.45% of the population is affected by symptoms caused by salivary calculi, though the estimated frequency including asymptomatic form may exceed 1% in adult population. Radiographs presenting a large portion of the maxillofacial region (panoramic radiography, computed tomography) could detect salivary calculi with high accuracy. The size of the sialoliths is usually less than 10 mm in diameter. Salivary calculi larger than 15 mm (considering the largest diameter) are classified as giant sialoliths and most of them are located in the submandibular gland or in its duct. Two unusually large submandibular salivary calculi cases are represented (diameters of 27 and 34 mm), whereas in one of the cases development and dimensional changes of the calculus are described via a seven years period. This case report represents diagnostic and therapeutic consequences in giant sialolithiasis and demonstrates possible differential diagnostic difficulties. Orv. Hetil., 2016, 157(49), 1967-1972.

Case report of giant sialolith (megalith) of the Wharton's duct.

Sialolithiasis is the most common disease of salivary glands. Its estimated frequency is 1.2% in the adult population. Sialoliths most commonly occur in the submandibular glands. The sublingual gland and minor salivary glands are rarely affected. The sialolith usually measures from 1 to <10 mm. Giant sialoliths are classified as those exceeding 15 mm in any one dimension. In literature, large sialoliths or megalith (> mm) of Wharton's duct have rarely been reported. This case report describes a patient presenting with an unusually large sialolith (megalith) of Wharton's duct, which was 37 mm ×16 mm in the size, the subsequent patient management, the etiology, diagnosis, and its treatment.

Rigid swelling of sublingual caruncle area due to the salivary gland duct obstruction by a sialolith

Sialolithiasis is the presence of calculus within the ductal system of a salivary gland. Among the diagnostic methods are inspection, palpation, checking the amount of saliva secreted and the identification of a sialolith. The authors present the case of a 37-year-old female patient with edema of the submandibular area and a bulging sublingual caruncle due to a calculus that obstructed the salivary gland ostium.

Rigid swelling of sublingual caruncle area due to the salivary gland duct obstruction by a sialolith.

Sialolithiasis is the presence of calculus within the ductal system of a salivary gland. Among the diagnostic methods are inspection, palpation, checking the amount of saliva secreted and the identification of a sialolith. The authors present the case of a 37-year-old female patient with edema of the submandibular area and a bulging sublingual caruncle due to a calculus that obstructed the salivary gland ostium.

Extracellular Ca(2+) sensing in salivary ductal cells.

Ca(2+) is secreted from the salivary acinar cells as an ionic constituent of primary saliva. Ions such as Na(+) and Cl(-) get reabsorbed whereas primary saliva flows through the salivary ductal system. Although earlier studies have shown that salivary [Ca(2+)] decreases as it flows down the ductal tree into the oral cavity, ductal reabsorption of Ca(2+) remains enigmatic. Here we report a potential role for the G protein-coupled receptor, calcium-sensing receptor (CSR), in the regulation of Ca(2+) reabsorption by salivary gland ducts. Our data show that CSR is present in the apical region of ductal cells where it is co-localized with transient receptor potential canonical 3 (TRPC3). CSR is activated in isolated salivary gland ducts as well as a ductal cell line (SMIE) by altering extracellular [Ca(2+)] or by aromatic amino acid, L-phenylalanine (L-Phe, endogenous component of saliva), as well as neomycin. CSR activation leads to Ca(2+) influx that, in polarized cells grown on a filter support, is initiated in the luminal region. We show that TRPC3 contributes to Ca(2+) entry triggered by CSR activation. Further, stimulation of CSR in SMIE cells enhances the CSR-TRPC3 association as well as surface expression of TRPC3. Together our findings suggest that CSR could serve as a Ca(2+) sensor in the luminal membrane of salivary gland ducts and regulate reabsorption of [Ca(2+)] from the saliva via TRPC3, thus contributing to maintenance of salivary [Ca(2+)]. CSR could therefore be a potentially important protective mechanism against formation of salivary gland stones (sialolithiasis) and infection (sialoadenitis).

Intraoral removal of a giant submandibular sialolith obstructing Wharton's duct: a case report.

Sialolithiasis is one of the most common pathologies of the submandibular gland; sialoliths account for about 80 percent of all salivary duct calculi. This report presents the unusual case of a large asymptomatic sialolith of the submandibular duct, initially diagnosed as a possible tumor. The giant sialolith was removed via an intraoral approach under local anesthesia. The etiology, pathogenesis, and management of such giant sialoliths are discussed.

Giant submandibular sialoliths: a report of two cases.

Sialolithiasis is the second most common disease of the salivary glands. Sialolithiasis accounts for the most common cause of salivary gland obstruction, leading to recurrent painful swelling of the involved gland, which often becomes worse while eating. Sialoliths can occur in any of the salivary glands but appear most frequently in the submandibular gland and its duct. Very few cases of giant submandibular sialoliths have been reported in the literature. This article reports two cases of unusually large submandibular sialoliths, with clinical and radiographical signs and management. The article also reviews the various diagnostic modalities and treatment options available.

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.

Causes, natural history, and incidence of salivary stones and obstructions.

Uncertainty about the causes and natural history of salivary stones (sialoliths) and other obstructions is being dispelled by clinical and experimental research. Sialoliths are now shown to be secondary to chronic obstructive sialadenitis. Microscopic stones (sialomicroliths) accumulate during secretory inactivity in normal salivary glands and produce atrophic foci by obstruction. Microbes ascend the main salivary duct during secretory inactivity and proliferate in atrophic foci and cause spreading inflammation, leading to inflammatory swelling and fibrosis that can compress large ducts. This leads to stagnation of secretory material rich in calcium that precipitates onto degenerating cellular membranes to form a sialolith.

Multiple, large sialoliths of the submandibular gland duct: a case report.

This paper reviews the major clinical and radiographic features of sialoliths and illustrates these with an unusual case of multiple sialoliths within the submandibular gland duct. The differential diagnosis of other calcific structures both within and outside the salivary gland that may mimic a sialolith is also presented.

[Ultrafast MR sialography: comparison of two coil systems including an alternative surface coil]. / Ultraschnelle MR-Sialografie: Intraindividueller Vergleich einer Oberflächenempfangs- mit einer konventionellen Kopf-Hals-Spule.

PURPOSE: To compare the visualization of salivary ducts by ultrafast magnetic resonance sialography (MR sialography) using an alternative surface coil and a conventional head-neck coil. MATERIALS AND METHODS: In 11 healthy volunteers, ultrafast MR sialography (single shot turbo spin echo; acquisition time, 2.8 s) was performed before and after oral application of a sialogogue. Each subject received examinations with both a bilateral surface coil (SC) and a conventional head-neck coil (HNC). The depiction of parotideal and submandibular duct systems was graded from 1 to 5 (5 = poor) by four independent radiologists. ANOVA served for statistical analysis of duct rankings, and interobserver variability was determined by Intraclass Correlation Coefficient (ICC). RESULTS: With an excellent ICC of 0.96, both coil systems offered symmetric visualization of salivary ducts, and the image quality increased after oral application of sialogogue (p < 0.001). In total, the overall rating was worse for SC than for HNC (2.13 +/- 1.24 vs. 1.45 +/- 0.65, p < 0.001). SC was especially inferior in depiction of submandibular and extraglandular duct components compared to HNC (p < 0.001). CONCLUSION: Most notably due to the reduced visualization of extraglandular and submandibular ducts, the specific surface coil used in this study was inferior in image quality and does not constitute a reasonable alternative to conventional coil systems.

Postenucleation orbital sialocele in a dog associated with prior parotid duct transposition.

An orbital sialocele developed in a dog following enucleation for protracted glaucoma. This eye had historically been treated for keratoconjunctivitis sicca by parotid duct transposition approximately 5 years previously, and the duct was ligated distally at the time of enucleation. One month following enucleation, the dog presented with a fluctuant conical-shaped swelling ventrolateral to the orbital socket. Surgical exploration revealed a dilated, fibrotic distal portion of the previously transposed parotid duct, and saliva, within the enucleated orbit. The distal portion of the duct and saliva-containing tissues from within the orbit were excised. The remaining proximal normal portion of the parotid duct was re-routed into the oral cavity. Clinicopathologic and histologic examination of the excised orbital contents and dilated portion of duct revealed a sterile sample of saliva and moderate chronic periductal fibrosis. At a 6-month re-evaluation there was no evidence of recurrence of the sialocele, and the parotid duct was functional.