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.

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.

Histological and chemical view on parotid duct sialolithiasis in the Slovakian warmblood mare.

The parotid duct has been reported to be the most common site of sialoliths in horses. In this case report, we described the first confirmed case of the equine sialolithiasis in Slovakia. The work was aimed to describe the transcutaneous approach to removing the sialolith, which manifested as a hard painless mass in the area of the maxillary cheek teeth, in a 14-year-old Slovakian warmblood mare. Pathological-anatomical and histological examination after extirpation confirmed the presence of parotid duct ectasia resulting from calculus. The mineral composition of the sialolith was determined with atomic absorption spectroscopy using X-ray powder diffraction. The sialolith was successfully extirpated transcutaneously, without complications or recurrence.

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).

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.

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.

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.

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.

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.

[Asymptomatic salivary gland stone of unusual size in the Wharton's duct]. / Wharton kanalinda semptom vermeyen dev tükürük bezi tasi.

A 44-year-old male patient presented with a complaint of dysphonia. On oropharynx examination, a polypoid mass was detected in the right vocal cord and firmness in the floor of the mouth, 3-4 cm in size. Computed tomography showed a stone in the right Wharton's duct. Saliva came out of the orifice of the Wharton's duct on palpation. The stone was removed transorally. Despite its huge size (30x20 mm), the patient had no complaint. No recurrence was seen during a follow-up period of 18 months with ultrasonography.

Parotid duct sialolithiasis in a patient with Down syndrome--case report.

Sialolithiasis is the phenomenon of blockage of the salivary glands. It frequently is associated with swelling, pain, and infection of the affected gland. Clinically, sialolithiasis manifests as an increase in the size of the affected gland and increased salivary secretion that results in pain during eating. It occurs mainly in the submandibular gland and less frequently in the parotid gland. This article presents a case involving a 23-year-old woman with Down syndrome who demonstrated sialolithiasis in the parotid duct gland. The sialolith was radiographed and removed surgically.

Clinicopathological study of myoepithelial sialadenitis and chronic sialadenitis (sialolithiasis).

To determine any overlap in pathological features between myoepithelial sialadenitis and chronic sialadenitis/sialolithiasis histological sections from 69 cases of myoepithelial sialadenitis (MESA) (n = 7) and chronic sialadenitis/sialolithiasis (n = 62) were reviewed over a 10 year period. Three of the cases with MESA contained calculi and four of those originally diagnosed as chronic sialadenitis/sialolithiasis showed epimyoepithelial island formation. The presence of calculi should not rule out a diagnosis of MESA, particularly in the parotid gland where calculi are uncommon; as the incidence of MESA may very well be underestimated and diagnosed as chronic sialadenitis, these patients, who are at increased risk of developing lymphoma, could be lost to follow up.

Salivary calculi: ultrastructural morphology and bacterial etiology.

Ultrastructural morphology of 16 salivary calculi was studied by means of transmission and scanning electron microscopy. The external surface was mostly globular or coarse, and on high magnifications and features could be divided into four main groups: a) amorphic calcified deposits covering extensive areas, b) other areas covered with crystals in a variety of arrangements, c) heavy accumulations of calcified rod-like and filamentouslike microorganisms, and d) platelet crystals in juxtaposition to calcified microorganisms in several areas. In most calculi the split area was found to be laminated. It is suggested that microorganisms have an important role in the formation and growth of salivary calculi.

The fine surface structure and composition of salivary calculi.

Salivary calculi were formed by a laminated mixture of inorganic and organic substancs. However, there was no regular pattern for the growth of stones, as laminal thickness and the arrangement of the laminal component were not uniform. Finger-like radial projections, longitudinal pavements or amorphous structures were seen on the fractured surface of the calculi. No inert foreign body or microorganism was found in the calculi. There were two modes of formation in calculous nucleus, one developed by maturation of a primitive core, the other formed in homogenous mineral mass. X-ray microanalysis proved that most salivary calculi contained chemical elements such as calcium, phosphorous, potassium, sodium, magnesium, chlorine, sulfur, manganese, chromium and aluminum. As far as we have examined, these elements were distributed with almost equal density throughout the calculus.

Large calculi of the submandibular salivary glands.

Salivary calculi occur in the submandibular and parotid glands, and their ducts, and occasionally reach a large size. However, little information is available on the composition of these giant stones. 2 cases are reported of unusually large calculi of the submandibular salivary glands. The glands were excised, and the results of chemical and infrared analysis of the calculi are presented.

Sialolithiasis of a Blandin's gland duct.

A case of sialolithiasis in a Blandin's gland duct is reported. Although minor salivary glands calculi are not uncommon, Blandin's glands are rare sites of occurrence. In this case, a calculus without decalcification is reported. Not only clinico-pathological features but also electron probe micro analyzer plus energy dispersive X-ray spectroscopy studies are presented. These studies reveal that phosphorus is contained in only the basophilic part of the calculus and sulphur in both the basophilic and the eosinophilic parts.