Removal of intraglandular calculi in Wharton's duct: clinical outcome and treatment algorithm.

The aim of this study was to propose a treatment strategy for intraglandular submandibular calculi based on calculus site. Seventy-three consecutive patients with impalpable intraglandular submandibular calculi were enrolled retrospectively. The calculi were classified as either post-hilar type, central type, or superficial type. Treatment approaches included transoral duct slitting (TDS), interventional basket retrieval (IBR), intraductal laser lithotripsy (ILL), and transcervical lithotomy (TCL). Complete calculus removal with gland preservation was achieved in 64 patients (87.7%). The success rate for post-hilar, central, and superficial calculi was 86.4% (51/59), 90.9% (10/11), and 100% (3/3), respectively. The treatment approach applied in patients with treatment success was TDS in 32 cases, IBR in 20, ILL in nine, and TCL in three. During follow-up (median 17.3 months), one patient experienced gland atrophy and three had ductal stenosis; the remaining 60 patients (93.8%, 60/64) had good clinical outcomes. In the eight failure cases operated by TDS, the deeply situated calculi could not be detached despite the parenchymal incision in five cases, while the procedure was ceased due to the patient's inability to cooperate in the other three cases. In the remaining failure case, the submandibular gland was sacrificed after calculus extraction via TCL. Application of the proposed treatment algorithm might help preserve gland function in patients with intraglandular submandibular calculi.

Salivary Mucinous Adenocarcinoma Masquerading as Chronic Submandibular Sialolithiasis.

A patient with chronic submandibular sialolithiasis underwent conservative treatment with appropriate imaging and multiple biopsies that continually revealed chronic inflammation. Due to continued symptoms, the patient underwent eventual excision and finaly pathology revealed salivary mucinous adenocarcinoma, which is a rare and poorly understood salivary malignancy. Persistent diagnostic workup and a high suspicion for salivary gland lesions is important for appropriate diagnosis and treatment. More attention and research on this specific entity can help future clinicians better diagnose and treat patients with a similar presentation. Laryngoscope, 134:2258-2261, 2024.

Venous malformation phleboliths mimicking submandibular sialadenitis in children.

To report one paediatric patient who presented with a rare venous vascular malformation as a mass in the left submandibular region with a clinical picture compatible with sialadenitis. Phleboliths are a specific feature of venous malformations due to venous stasis and may mimic sialoliths on various imaging modalities. Thus venous malformations are often misdiagnosed as sialadenitis due to sialolithiasis. Sialoliths are extremely rare in paediatric patients. In an early adolescent presenting with a submandibular mass and suspected sialadenitis arising from sialoliths, a detailed history, clinical examination and careful review of the radiological findings will allow the diagnosis of venous vascular malformation and provide the complete surgical resection. Our patient was initially referred with a presumed diagnosis of submandibular sialadenitis, and instead a venous malformation with phlebolith was diagnosed.

[Lithiasic salivary gland pathologies]. / Pathologies lithiasiques des glandes salivaires.

LITHIASIC SALIVARY GLAND PATHOLOGIES. Sialolithiasis is the most frequent pathology affecting the salivary glands. Almost 80% of these lithiasis affect the submandibular gland, in the Wharton duct whilst 20% occur in the parotid gland in the Stenon duct. The diagnosis is based on typical patient interrogation and clinical symptoms (intermittent facial swelling associated with eating, sialadenitis) and/or on paraclinical examination. Ultrasound echography is often prescribed, but CT-scan and sialo-MRI offer a more precise preoperative assessment. Minimally-invasive gland-preserving techniques such as sialendoscopy (combined or not with transoral duct surgery) are nowadays considered to be the method of first choice. These methods reach success rates above 80% and significantly reduce surgical complications and hospital stay.

PATHOLOGIES LITHIASIQUES DES GLANDES SALIVAIRES. Parmi les pathologies des glandes salivaires, les lithiases des canaux salivaires sont les plus fréquentes. Elles sont le plus souvent retrouvées dans les glandes sous-maxillaires au niveau des canaux de Wharton (80 %), et plus rarement dans les glandes parotides au niveau des canaux de Sténon (20 %). Le diagnostic repose sur l'interrogatoire et la clinique, qui peuvent être évocateurs (hernie ou coliques salivaires, sialite aiguë), et sur l'imagerie. L'échographie est un examen diagnostique très accessible, mais le scanner et la sialo-IRM permettent une évaluation préopératoire plus précise. Le traitement repose en première intention sur des traitements chirurgicaux conservateurs et mini-invasifs tels que la sialendoscopie, associée ou non à une approche transorale, qui sont efficaces dans 80 % des cas et permettent de réduire les complications postopératoires et la durée d'hospitalisation.

A comparative study of three-dimensional cone-beam CT sialography and MR sialography for the detection of non-tumorous salivary pathologies.

BACKGROUND: Imaging of the salivary ductal system is relevant prior to an endoscopic or a surgical procedure. Various imaging modalities can be used for this purpose. The aim of this study was to compare the diagnostic capability of three-dimensional (3D)-cone-beam computed tomography (CBCT) sialography versus magnetic resonance (MR) sialography in non-tumorous salivary pathologies. METHODS: This prospective, monocenter, pilot study compared both imaging modalities in 46 patients (mean age 50.1 ± 14.9 years) referred for salivary symptoms. The analyses were performed by two independent radiologists and referred to identification of a salivary disease including sialolithiasis, stenosis, or dilatation (primary endpoint). The location and size of an abnormality, the last branch of division of the salivary duct that can be visualized, potential complications, and exposure parameters were also collected (secondary endpoints). RESULTS: Salivary symptoms involved both the submandibular (60.9%) and parotid (39.1%) glands. Sialolithiasis, dilatations, and stenosis were observed in 24, 25, and 9 patients, respectively, with no statistical differences observed between the two imaging modalities in terms of lesion identification (p1 = 0.66, p2 = 0.63, and p3 = 0.24, respectively). The inter-observer agreement was perfect (> 0.90) for lesion identification. MR sialography outperformed 3D-CBCT sialography for visualization of salivary stones and dilatations, as evidenced by higher positive percent agreement (sensitivity) of 0.90 [95% CI 0.70-0.98] vs. 0.82 [95% CI 0.61-0.93], and 0.84 [95% CI 0.62-0.94] vs. 0.70 [95% CI 0.49-0.84], respectively. For the identification of stenosis, the same low positive percent agreement was obtained with both procedures (0.20 [95% CI 0.01-0.62]). There was a good concordance for the location of a stone (Kappa coefficient of 0.62). Catheterization failure was observed in two patients by 3D-CBCT sialography. CONCLUSIONS: Both imaging procedures warrant being part of the diagnostic arsenal of non-tumorous salivary pathologies. However, MR sialography may be more effective than 3D-CBCT sialography for the identification of sialolithiasis and ductal dilatations. TRIAL REGISTRATION: NCT02883140.

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.

Identification of biological components for sialolith formation organized in circular multi-layers.

According to the previous studies of sialolithiasis reported so far, this study is aimed to identify the biological components of sialolith, which show different ultrastructures and chemical compositions from other stones, cholelith and urolith. Twenty-two specimens obtained from 20 patients were examined histologically, and analyzed with micro-CT, scanning electron microscopy (SEM), energy dispersive X-ray spectroscopy (EDS), and transmission electron microscopy (TEM). All sialoliths (n = 22) observed in this study showed a central nidus, which was filled with organoid matrix admixed with exosome vesicles, loose calcium apatite crystals, and many bacteria. The micro-CT and SEM observation clearly defined a single or multiple central nidus(es) encircled by highly calcified compact zone. The circular compact zone showed a band-like calcification, about 1-3 mm in thickness, and usually located between the central nidus and the peripheral multilayer zone. But some sialoliths (n = 5) showed severe erosion of compact zone by expanding multilayered zone depending on the level of calcification and inflammation in sialolith. By observing TEM images, many exosome vesicles and degraded cytoplasmic organelles were found in the central nidus, and some epithelial cells were also found in the calcified matrix of peripheral multilayer zone. Particularly, EDS analysis indicated the highest Ca/P ratio in the intermediate compact zone (1.77), and followed by the central nidus area (1.39) and the peripheral multilayer zone (0.87). Taken together, these data suggest that the central nidus containing many inflammatory exosomes and degraded cytoplasmic organelles has a potential to induce a band-like calcification of compact zone, and followed by the additional multilayer deposition of exfoliated salivary epithelial cells as well as salivary materials. Thereby, the calcium apatite-based sialolith is gradually growing in its volume size, and eventually obstructs the salivary flow and provides a site for the bacterial infection.

Submandibular sialolith migration.

A middle-aged male Caucasian had rejected previous offers of surgery for submandibular gland removal in the past due to concerns about surgical complications. He presented with a month's history of submandibular swelling and severe pain, which impeded his ability to eat. Prior to admission, he had been experiencing intermittent sialadenitis for several months. Cross-sectional imaging demonstrated a 16×12 mm migratory sialolith, located superficial to the right submandibular gland within a large loculated abscess. The patient underwent an incision and drainage of the abscess under general anaesthetic and the sialolith was expressed. He was discharged home with oral antibiotics and was followed up as an outpatient. This case serves to highlight a rare complication of chronic sialolithiasis.

A comparative study of three-dimensional cone beam computed tomographic sialography and ultrasonography in the detection of non-tumoral salivary duct diseases.

OBJECTIVES: To compare the overall diagnostic outcomes of 3D-CBCT sialography and ultrasonography (US) in the detection of sialolithiasis, ductal dilatation, and ductal stenosis. METHODS: This retrospective monocentric study compared the two imaging modalities carried out in the same patients referred for salivary symptoms of the parotid and submandibular glands. The primary endpoint was the capacity of the imaging procedure to diagnose a lesion. The secondary objectives were the detection rates according to the type of lesion, analysis of the causes of failure, and the parameters of radiation exposure and safety (for 3D-CBCT sialography). RESULTS: Of the 236 patients who received a 3D-CBCT sialography in our institution, 157 were ultimately included in the per-protocol analysis. 3D-CBCT sialography allowed detection of ductal lesions in 113 patients versus 86 with US. The two imaging modalities yielded congruent interpretations in 104 out of 157 subjects (66.2%). Higher sensitivity and negative predictive value were observed with 3D-CBCT sialography compared with US, irrespective of the lesions studied: 0.85 vs 0.65 and 0.70 vs 0.44, respectively. Regarding the sialolithiasis, both 3D-CBCT sialography and US allowed identification of lesions with high sensitivity and negative predictive value (0.80 vs 0.75 and 0.88 vs 0.78, respectively). CONCLUSIONS: US remains the first-line examination for exploration of the salivary lesions. 3D-CBCT sialography is an alternative in case of inconclusive US, and prior to any endoscopic procedure.

Treatment outcomes of sialendoscopy for submandibular gland sialolithiasis.

OBJECTIVE: Sialendoscopy is a procedure used to remove salivary stones intraorally using a sialendoscope. In this study, we identified treatment outcomes of sialendoscopic surgery and identified predictive factors for successful stone removal by sialendoscopy alone. METHODS: We assembled the medical records of 144 patients who underwent sialendoscopic surgery for submandibular gland sialolithiasis at the Department of Otolaryngology-Head and Neck Surgery, Asahikawa Medical University, from October 2010 to November 2021, and collected patient backgrounds, medical condition, perioperative factors including operation method and complications, postoperative course, and stone constituents from a clinical laboratory testing company. RESULTS: Submandibular gland stones were successfully removed using sialendoscopy in 58 patients (40%). In multivariate analysis, location, major axis, and mobility of the stones were independent factors for successful removal. In receiver operating characteristic analysis, <7.5 mm of a major axis may be used as a measuring standard for successful removal. Removal of parenchymal stones is prone to involve prolonged operation times, increased postoperative complications, and development of retained stones. The stones mainly consisted of calcium phosphate and protein, with content percentages ranging from 0 to 98% (median 37%) and from 0 to 100% (median 63%), respectively. The percentage of calcium phosphate was negatively correlated with the number of floating stones and successful stone removal. CONCLUSION: Sialendoscopy is an aesthetically attractive treatment for sialolithiasis that avoids cervical incisions. The present results showed not only known but also new predictive factors for the successful removal of stones (<7.5 mm) and percentage of calcium phosphate. Moreover, our results suggest that careful consideration is required regarding the indication of sialendoscopic surgery in patients with parenchymal stones.

Huge sialolith of the submandibular gland: a case report and review of literature.

The formation of stone in the ductal system of the salivary gland is termed sialolithiasis, with the submandibular gland being the most commonly affected. The precise aetiology is unknown but certain factors peculiar to the submandibular gland accounts for its likelihood of developing a calculous disease. Stones are classified based on their dimension, and may be silent or present with symptoms attributable to the size, location and complications. Here, a 50-year-old female who presented with a painless swelling in the left submandibular region that had grown slowly over the previous year, is reported. Following clinical and radiologic evaluation, left submandibular stone disease was preoperatively diagnosed. The patient underwent sialoadenectomy with transient palsy of the marginal mandibular branch of the facial nerve. Histopathology confirmed sialolith (2.7 cm) with severe squamous metaplasia of the duct. This presentation demonstrates some peculiar features attributable to the size, vertical orientation and location of the stone, in addition to the compression of the gland, thick fibrous capsule and significant squamous metaplasia of the duct. These findings require further evaluation for optimal treatment in view of the emerging trends for managing sialolithiasis.

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.

Sialendoscopy-assisted combined approach for parotid sialolithotomy: Our long-term experience.

OBJECTIVES: To assess the long-term outcome of sialendoscopy-assisted combined approach for parotid sialolithotomy with gland preservation. PATIENTS AND METHODS: A retrospective study of patients treated with a combined sialendoscopic and open approach was conducted between 2011 and 2020. Demographic data of patients such as operative technique, stone size, stone location, complications, and symptom relief were collected. Patients were followed up via clinical examination and questionnaires. RESULTS: Seventy-four patients were included and underwent endoscopy-assisted combined operations for the removal of 98 parotid stones. Of the 98 stones, 92(94%) stones were completely removed and 6(6%) were partially removed. At a mean follow-up of 47.1 ± 35 months, 65 of 74 patients (88%) achieved long-term success. Patients with stone incomplete removal were significantly more often to develop the recurrence of obstructive symptoms (p = 0.000) There were no cases of facial nerve injury or fistula formation. Gland function was preserved in 73 of 74 patients (99%). CONCLUSIONS: The combined approach for parotid stones is a safe and gland-preserving alternative to parotidectomy. The techniques described here show high success rates and good long-term results, and they avoided the need for gland resection in >95% of cases.

Sialography: a pictorial review.

Non-tumour inflammatory and obstructive salivary gland pathologies such as sialadenitis, sialolithiasis, sialadenosis, ductal strictures, etc. require precise radiological evaluation and mapping of salivary gland ductal system for better treatment outcome. Conventional sialography is considered as a useful and reliable technique in evaluation of salivary glands especially intrinsic and acquired abnormalities involving the ductal system and is useful for detection of non-radiopaque sialoliths which are invisible on routine plain radiographs. Primarily sialography is used as a diagnostic tool, additionally it plays an important therapeutic role as salivary gland lavage in cases of recurrent salivary gland infections and in obstructive salivary gland disorders by helping in clearance of mucous plugs or small sialoliths within the ducts. Recently, diagnostic performance of computed tomography (CT) sialography is being explored and has been reported to have high sensitivity in detection of small sialoliths and allows differentiation of sialoliths from other calcifications in glandular ductal system. Multiplanar three dimensional (3D) reconstructed CT images have been reported to play a key role in determination of anatomical location or extent of salivary gland disease without superimposition or distortion of structures. This review aims to discuss the disease specific applications of sialography and CT Sialography in particular for visualization of salivary gland disorders.

Sialolithiasis: mineralogical composition, crystalline structure, calculus site, and epidemiological features.

The purpose of this paper was to describe the characteristics of salivary calculi and their relationship to epidemiological factors, through a cross-sectional study. We analysed 100 calculi obtained in 2017-2021. Patient data including age, time since onset of symptoms, gland involved, and site of location in the salivary system were studied. The calculi were studied to determine their morphological features using scanning electron microscopy and energy dispersive plain radiographic analysis. Most of the calculi had formed in the submandibular gland (SG) (82%). The mean age of patients at onset was 45.83 years; patients presenting parotid gland (PG) stones were somewhat older (p = 0.031). The mean time since the onset of symptoms was longer in PG calculi (p = 0.038). The most common lithiasis site was the main duct (74%), followed by the hilum (22%). Hilar stones were the largest (p < 0.05) and heaviest (p = 0.028). Octacalcium phosphate (OCP) was the most common crystalline phase (Cp) founded, followed by hydroxyapatite (HA) and whitlockite (WH). Specifically, OCP had a higher presence in PG calculi (p = 0.029) and WH was the most common phase in SG calculi (p = 0.017). The most prevalent site of lithiasis was the main duct, and the largest and heaviest calculi were found in the SG. PG stones were associated with a longer history of symptoms and older age. OCP was the most frequent Cp of the calculi studied, and the main Cp in PG stones. WH was the predominant Cp in SG stones. The Cp of the calculi was not influenced by location, patient age, or time of symptoms.

Giant submandibular sialolith: a case report.

Introduction: Salivary glands lithiasis (Sialolithiasis) is defined as calcified concretions in the salivary glands. Most common localization is in submandibular gland. Usually, submandibular stones are mainly located in Wharton's duct, whereas parotid stones are more often located in the gland parenchyma. Sialoliths are usually 5-10 mm in size, and stones more than 10 mm are unusual sizes. Exact etiology of sialolith formation is still unknown. Case Report: We discuss a case of a 70-year-old patient, presenting painful swelling and a giant submandibular gland sialolith successfully treated with open surgery. Conclusions: A careful anamnesis and physical examination of the patient are important in the diagnosis of sialolithiasis. In addition, several imaging techniques, such as panoramic X-rays and Ultrasound, can be applied. The management can be both medical and surgical.

Utility of Ultrasonography for Diagnosis of Salivary Gland Sialolithiasis: A Meta-Analysis.

OBJECTIVES: We hypothesized that ultrasonography for salivary gland stone detection would have a diagnostic accuracy similar to that confirmed by sialendoscopy, sialography, or surgery. Therefore, we evaluated the diagnostic characteristics of ultrasonography in terms of submandibular and parotid stone detection compared to confirmatory methods. METHODS: We searched PubMed, Embase, the Web of Science, SCOPUS, and the Cochrane database to October 31, 2021. The risk of bias was evaluated using the QADAS-2 tool. RESULTS: Ten studies involving 1393 patients were included in the analysis. The diagnostic odds ratio of ultrasonography was 162.6013 (95% confidence interval [CI] [53.9883; 489.7208] and I2 value 81.0%). The area under the summary receiver operating characteristic curve was 0.963. The sensitivity, specificity, negative predictive value, and positive predictive value were 0.8992 (95% CI [0.8534; 0.9318]; I2  = 79.9%), 0.9664 (95% CI [0.9290; 0.9844], I2  = 65.6%), 0.8076 (95% CI [0.7256; 0.8694]; I2  = 80.4%), and 0.9853 (95% CI [0.9629; 0.9943]; I2  = 77.4%), respectively. However, high-level among-study heterogeneity (I2 ≥ 50%) was evident, attributable to the inclusion of different glands. On subgroup analysis, significant differences in the negative predictive values (parotid gland only [0.9392], submandibular gland only [0.6718], and parotid and submandibular glands [0.8105]) were apparent. We found no significant among-study difference in the sensitivity, specificity, positive predictive value, or diagnostic odds ratio (P > .05). CONCLUSION: Ultrasonography usefully detects submandibular and parotid gland stones. Ultrasonography of the parotid gland was associated with the highest diagnostic accuracy, but further clinical studies are needed. LEVEL OF EVIDENCE: NA Laryngoscope, 132:1785-1791, 2022.