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.

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.

Sialendoscopy in treatment of obstructive sialadenitis.

OBJECTIVES: Sialendoscopy is a relatively new mini-invasive method that allows direct visualisation and intervention in the salivary gland ductal system. The aim of the study was to evaluate the results of sialendoscopy in the treatment of obstructive sialadenitis. MATERIALS AND METHODS: This is a 15-year retrospective study analysing the treatment results of patients treated in the period of 2007-2022 at the Department of Oral and Maxillofacial Surgery, Comenius University Bratislava, Slovakia. RESULTS: The total number of performed sialendoscopies was 70, of which 44 (62.9 %) were performed on the submandibular gland and 26 (37.1 %) on the parotid gland; 46 procedures (65.7 %) were performed via natural ductal system opening without the need for surgical assistance while 24 sialendoscopies (34.3 %) required surgical assistance. The most frequent perioperative findings were the sialoliths (37) in quantities ranging from one to four. Non-calculi pathologies (23) included mucous plugs, strictures, plaque, erythema or foreign bodies. No pathology was found on 10 sialendoscopies. In 82 % (n = 55) of patients, sialendoscopy prevented the salivary gland from being excised. In 18 % (n = 12) of cases, sialendoscopy findings indicated that salivary gland excision was needed. CONCLUSION: The study acknowledges the significant benefit of sialendoscopy in the treatment of obstructive sialadenitis (Tab. 3, Fig. 6, Ref. 39). Text in PDF www.elis.sk Keywords: sialendoscopy, sialadenitis, duct obstruction, sialolith, minimally invasive surgery.

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.

Ablation of TRPC3 disrupts Ca2+ signaling in salivary ductal cells and promotes sialolithiasis.

Clinical studies and structural analyses of salivary stones strongly suggest a linkage between higher saliva calcium (Ca2+) and salivary stone formation, sialolithiasis; however, the process and the mechanism leading to Ca2+ overload during sialolithiasis is not well understood. Here, we show that TRPC3 null (-/-) mice presented with a reduction in Ca2+ entry and current in ductal cells with higher saliva [Ca2+] suggesting diminished transepithelial Ca2+ flux across the salivary ductal cells, leaving more Ca2+ in ductal fluid. Significantly, we found that TRPC3 was expressed in mice and human salivary ductal cells, while intraductal stones were detected in both mice (TRPC3-/-) and patient (sialolithiasis) salivary glands. To identify the mechanism, we found that TRPC3 was crucial in preventing the expression of calcification genes (BMP2/6, Runx2) in ductal cells which may be due to higher extracellular Ca2+ in SMG tissues. Similarly, inflammatory (IL6, NLRP3), fibrotic (FN1, TGFß1) and apoptotic (Bax1/Bcl2) markers were also elevated, suggesting that the loss of TRPC3 induces genetic changes that leads to salivary gland cell death and induction of inflammatory response. Overall, ablation of TRPC3-/- leads to higher saliva [Ca2+], along with elevated detrimental gene expressions, altogether contributing to salivary gland stone formation.

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.

The effectiveness and safety of sialendoscopy for sialolithiasis in children: a systematic review and meta-analysis.

PURPOSE: Sialendoscopy is a new, minimally invasive method that is becoming increasingly more popular than traditional methods for the treatment of sialolithiasis. In this systematic review and meta-analysis, the effectiveness and safety of this method in children with sialolithiasis are investigated. METHODS: Inclusion criteria were children with sialolithiasis. The information sources were databases MEDLINE and PubMed Central (through PubMed), ScienceDirect, Cochrane Central Register of Controlled Trials (CENTRAL), citation indexes Scopus and Google Scholar, trial registries, and "gray literature". The last search was performed on September 18, 2022. The risk of bias in included studies was assessed using ROBINS-I tool (Risk Of Bias In Non-randomized Studies of Interventions). The pooled proportion of weighted means was calculated for the quantitative synthesis of available data. RESULTS: The effectiveness of the method was estimated at 95.5% (95% CI 89.8-99.3%), from 13 studies including 133 cases. The safety was estimated at 97.2% (95% CI 91.8-100%), from 10 studies including 113 cases. CONCLUSION: The limitations of this study, briefly summarized, are the small number of included studies, the fact that they are mostly retrospective, the difficult application of the guidelines suggested by the PRISMA statement (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) and Cochrane handbook due to the nature of this specific condition and intervention, and the difficulty in assessing reporting bias. The results of the current study indicate that sialendoscopy is an effective and safe method for the treatment of sialolithiasis in children and should be implemented in daily clinical practice.

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.

[Recent progress in the treatment of intractable sialolithiasis].

Sialolithiasis occurs in approximately 0.45% to 1.20% of the general population. The typical clinical symptom manifests as a painful swelling of the affected glands after a meal or upon salivary stimulation, which extremely affects the life quality of the patients. With the development of sialendoscopy and lithotripsy, most sialoliths can be successfully removed with preservation of the gland. However, sialoliths in the deep hilar-parenchymal submandibular ducts and impacted parotid stones located in the proximal ducts continue to pose great challenges. Our research center for salivary gland diseases (in Peking University School and Hospital of Stomatology) has used sialendoscopy for 17 years and treated >2 000 patients with salivary gland calculi. The success rate was approximately 92% for submandibular gland calculi and 95% for parotid calculi. A variety of minimally invasive surgical techniques have been applied and developed, which add substantial improvements in the treatment of refractory sialolithiasis. Further, the radiographic positioning criteria and treatment strategy are proposed for these intractable stones. Most of the hilar-parenchymal submandibular stones are successfully removed by a transoral approach, including transoral duct slitting and intraductal basket grasping, while a small portion of superficial stones can be removed by a mini-incision in submandibular area. Impacted stones located in the distal third of parotid gland ducts are removed via "peri-ostium incision", which is applied to avoid a cicatricial stenosis from a direct ostium incision. Impacted parotid stones located in the middle and proximal third of the Stensen's duct are removed via a direct mini-incision or a peri-auricular flap. A direct transcutaneous mini-incision is commonly performed under local anesthesia with an imperceptible scar, and is indicated for most of impacted stones located in the middle third, hilum and intraglandular ducts. By contrast, a peri-auricular flap is performed under general anesthesia with relatively larger operational injury of the gland parenchyma, and should be best reserved for deeper intraglandular stones. Laser lithotripsy has been applied in the treatment of sialolithiasis in the past decade, and holmium ∶YAG laser is reported to have the best therapeutic effects. During the past 3 years, our research group has performed laser lithotripsy for a few cases with intractable salivary stones. From our experiences, withdrawal of the endoscopic tip 0.5-1.0 cm away from the extremity of the laser fiber, consistent saline irrigation, and careful monitoring of gland swelling are of vital importance for avoidance of injuries of the ductal wall and the vulnerable endoscope lens during lithotripsy. Larger calculi require multiple treatment procedures. The risk of ductal stenosis can be alleviated by endoscopic dilation. In summary, appropriate use of various endoscopy-assisted lithotomy helps preserve the gland function in most of the patients with refractory sialolithiasis. Further studies are needed in the following aspects: Transcervical removal of intraglandular submandibular stones, intraductal laser lithotripsy of impacted parotid stones and deep submandibular stones, evaluation of long-term postoperative function of the affected gland, et al.

Non-neoplastic salivary gland diseases in children: a 10-year review at a tertiary center.

OBJECTIVES: Non-neoplastic salivary gland diseases are rare in children. This paper aims to present the spectrum of encountered non-neoplastic salivary gland diseases at a tertiary center by describing the demographics, clinical characteristics, and outcomes in this patient population. METHODS: A review of electronic medical records was performed from 2010 until 2020. Relevant data were retrieved and charted according to the type of salivary gland disease. A comparison between diseases was made for demographics, presenting symptoms, treatment, and outcomes. RESULTS: Fifty patients with 11 different non-neoplastic salivary gland diseases were identified. Sialolithiasis was the most prevalent condition (12/50), with 83% localized in the submandibular gland. In contrast to gender, age, and symptoms, the location of pathology was significantly associated with the diagnosis (p < 0.001). In patients with sialolithiasis, a hybrid procedure (combined endoscopy and lithotomy) resulted in 100% resolution of symptoms. For (plunging) ranula, marsupialisation had a relative risk of recurrence of 9.6 compared to (partial) extirpation of the sublingual gland. CONCLUSIONS: Children with salivary gland diseases present with overlapping symptoms, making clinical diagnosis challenging. The present study may aid physicians and specialists in diagnosing the most prevalent conditions in children. Although no gold standards exist for their treatment, hybrid procedures (sialolithiasis) and subglingual gland extirpation (ranula) showed superior results over alternatives.

Evaluation of patients' quality of life using novel "Multidisciplinary Salivary Gland Society" questionnaire in minimally invasive management of obstructive sialadenitis.

OBJECTIVE: The introduction of minimally invasive surgical procedures has significantly reduced the rate of major salivary gland removal. This study aimed to assess these procedures in relieving patients' complaints and their impact on quality of life using the newly introduced Multidisciplinary Salivary Gland Society questionnaire. STUDY DESIGN: Between January 2021 and October 2021, a case series was treated with minimally invasive surgical procedures. Data such as stone size, number and location, surgical approach, symptoms relief, and improvement of quality of life were collected and analyzed. Thirty-six patients were included (22 males and 14 females); 28 had stones in the submandibular gland and 8 in the parotid gland with largest stone diameter ranging from 0.2 to 2.8 cm. The approach was selected according to stone size and location, so 17 patients were treated with sialendoscopy (alone or using a combined approach), and 19 patients were treated without sialendoscopy using a transoral approach. RESULTS: After a 6-month follow-up, the questionnaire scores showed a statistically significant difference in comparison to preoperative ones. CONCLUSIONS: Minimally invasive surgical procedures present an excellent option for sialolithotomy, after which the relief of symptoms, restoration of gland function, and a significant improvement in quality of life are achieved.

Reliable and safe salivary duct repositioning technique with partial transection and intraductal stenting: Technical notes.

Salivary duct repositioning is often performed after excision of malignant tumors or removal of sialoliths to maintain salivary function and minimize the risk of swelling or pain due to the obstruction of the gland. However, there is risk of intraoperative tissue damage due to traumatic manipulation, leading to stenosis; in addition, the small diameter of the duct also renders this procedure difficult. Recently, we improved our method of salivary duct repositioning as follows. In the first technique, partial transection is made on the lateral wall of the duct ligated with thread at the end. Pulling the thread provides a view of the lumen, and appropriate tension enables a reliable and non-invasive procedure without requiring the operator to grasp the edge of the duct directly. When the diameter of the duct is small, intraductal stenting, the second technique, can be combined with the former technique by probe insertion to expand the lumen. Our approach is technically easy and simple which can be accepted by any clinicians and it could also be a promising technique that can serve as a less invasive and effective treatment.

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.

Prognostic Factors for the Outcome of Salivary Gland Holmium: YAG Laser Intraductal Lithotripsy.

OBJECTIVES: Holmium: YAG laser has gained its popularity throughout the years and is used to treat sialolithiasis, which helps to overcome the limitations of traditional sialendoscopic lithotripsy for larger-sized salivary stones. However, little information is available regarding factors predicting the success rate of Holmium: YAG laser intraductal lithotripsy. The purpose of this study is to investigate the factors affecting the success rates of Holmium: YAG laser lithotripsy for salivary stones treatment in a tertiary care hospital. METHODS: A retrospective study conducted in patients receiving sialolithiasis surgery under sialendoscopy from May 2013 to March 2015 at Mackay Memorial Hospital, Taiwan. Data on various factors, including patients' age, gender, glands, size of largest stone, multiple stones (≥2 stones), location of the stone (distal duct, middle duct, proximal duct, and hilum), and operative time. The success of the surgery defined as patients without any complaints such as swelling or tenderness. Logistic regression and Fisher exact tests were employed to examine these factors on the success rate. RESULTS: Fifty-four patients who received sialendoscopy surgery with a mean age of 35.74 years old recruited. Logistic regression identified the operation time exceeding 210 minutes showed 23.497 folds higher odd ratio of having a result of operation failure (P < .05). CONCLUSION: The prolonged operation time is the sole independent factor affecting the successful outcome for salivary gland intraductal laser lithotripsy. We recommend operative time be no more than 210 minutes to increase the success rate in salivary gland Holmium: YAG laser intraductal lithotripsy.

Symptom Outcomes After Sialendoscopy-Assisted Salivary Duct Surgery: A Prospective 6-Year Study.

OBJECTIVE: The objective is to evaluate the long-term impact of sialendoscopic-assisted salivary duct surgery (SASDS) on sialadenitis symptoms using the Chronic Obstructive Sialadenitis Symptoms (COSS) questionnaire. METHODS: The COSS questionnaire, which scores symptoms on a 0-100 scale, was administered prospectively to adult patients pre-operatively, 1-year, and 6-years post-operatively. We examined COSS scores and categories representing complete (<10), partial (10-25), and no (>25) resolution of symptoms with attention to factors significantly associated with incomplete resolution (≥10). RESULTS: Approximately 6-years after SASDS, 111 patients reported scores for 128 symptomatic glands (72 with sialolithiasis, 56 without sialolithiasis). For glands with sialolithiasis, the median COSS score pre-SASDS was 27.5 (interquartile range [IQR]: 13.5-43), which was significantly reduced to 1.0 (IQR: 0-5.5) at 1-year and 1.5 (IQR 0-5) at 6-years postoperatively. Glands without sialolithiasis had a median COSS score of 40.5 (IQR: 23-52.5) preoperatively, that significantly reduced to 13.5 (IQR 5-21) at 1-year and 14 (IQR 6.5-25.5) at 6-years post-operatively. There was no significant difference in scores from 1- to 6-years. Intraoperative presence of sialolith, absence of stenosis, stenosis in the distal duct, and submandibular gland involvement were significant predictors of complete resolution of symptoms at 6-years. CONCLUSION: Approximately 6-years after SASDS for sialadenitis, the majority of patients have durable symptom improvement. The sialolithiasis group had higher rates of complete resolution compared to the non-sialolithiasis group. Presence of ductal stenosis on sialendoscopy, non-distal stenoses, and parotid gland involvement were risk factors for persistent long-term sialadenitis symptoms. Laryngoscope, 133:792-800, 2023.

Comparison of two transcutaneous approaches for the removal of impacted parotid stones.

The aim of this study was to comparatively evaluate the indications and treatment outcomes of two transcutaneous approaches for the removal of impacted parotid stones. Sixty-eight consecutive patients with impacted parotid stones underwent endoscopy-assisted lithotomy via a direct mini-incision or a peri-auricular flap. Clinical safety and outcomes were evaluated. Complete stone extraction was achieved in all patients. In the mini-incision group (52 patients), the stones were in the middle third of the main duct in 31 patients, at the hilum in 16, and in the intraglandular duct in five. In the flap group (16 patients), they were in the middle third of the main duct in one patient, at the hilum in seven, and in the intraglandular duct in eight. Salivary fistula occurred in five mini-incision group patients (9.6%) and four flap group patients (25%). The clinical outcome in the mini-incision group (47 patients, median 25 months of follow-up) was good in 28 patients, fair in 13, and poor in six (12.8%). The clinical outcome in the flap group (16 patients, median 84 months of follow-up) was good in nine patients, fair in five, and poor in two (12.5%). The direct mini-incision approach was found to be safe and effective for impacted stones in the middle third, hilum, and proximal third of the main duct, while the peri-auricular approach would be best reserved for deeper intraglandular stones.