This case report details a complex case of parotid gland sialolithiasis with stones adherent to the facial nerve, a scenario that presents a significant surgical challenge. Traditional sialendoscopy failed to address the condition in a 23-year-old female patient, leading to the adoption of a combined endoscopic transcutaneous approach. This method successfully resolved the condition without intraoperative complications, maintaining intact facial nerve function postoperatively. The case emphasizes the importance of individualized surgical strategy and expert technique in advanced parotid surgery, advocating this approach for similarly complex sialolithiasis cases.

BACKGROUND: Parotid gland agenesis is a rare, congenital, usually asymptomatic disorder. Until now, only 24 cases with unilateral, incidentally found, parotid gland agenesis have been described. Here, we present the first reported case of an ipsilateral preauricular neoplasm in a patient with unilateral parotid gland agenesis. During surgery, the position of the greater auricular- and facial nerves was documented. Furthermore, we performed the first sialendoscopy for this rare disorder to assess the number of duct branches, which might be indicative of the abundance of parotid tissue. Moreover, we looked for sialendoscopic characteristic features that could aid in identifying these patients in the ambulatory setting.
METHODS: A 50-year-old Greek man presented with a painless, slowly enlarging mass in the right parotid space. Magnetic resonance imaging revealed a complete absence of the right parotid gland without accessory parotid tissue. The right parotid gland was replaced by fatty tissue and the radiologist suggested a benign parotid tumor. Fine needle aspiration was indicative of a reactive lymph node. Sialendoscopy revealed only two branches within the right parotid duct. Surgical resection was performed through a conventional lateral parotidectomy. This revealed typical anatomic position of the greater auricular- and facial nerves despite the parotid tissue agenesis. Histopathology revealed a small lymphocytic lymphoma.
CONCLUSIONS: Surgeons should feel confident to resect tumors of the parotid space in patients with parotid gland agenesis. Reduced branching observed during sialendoscopy might indicate parotid gland agenesis. Physicians should be even more cautious than usual with the watch and wait strategy in patients with tumors of parotid gland agenesis, since the probability of a tumor being a benign salivary gland tumor might be lower than usual.

Juvenile recurrent parotitis (JRP) is characterised by recurrent episodes of painful parotid swelling in children. JRP is the second most common cause of parotitis in childhood, behind only paramyxovirus. The prevention of recurrent attacks represents the most dramatic and serious aspect of this pathology. Since 2004, different authors have evaluated sialendoscopy for the diagnostic and therapeutic management of JRP. In this paper, we share our clinical experience of the use of sialendoscopy for the treatment of JRP. We document with video sialendoscopy the glandular pathology in four children with a mean age of 11.5 years, who had suffered from 3-6 episodes/year of inflammation prior to treatment. The use of sialendoscopy in our patients was effective in preventing recurrences. For the first time, the videosialendoscopy of a series of children diagnosed with JRP is documented in the literature.

La recidiva della patologia ostruttiva salivare dopo scialoendoscopia. Una revisione narrativa della letteratura.
Negli ultimi 20 anni, la scialoendoscopia interventistica è diventata la terapia di scelta dei disordini ostruttivi salivari, contribuendo ad una riduzione significativa dei pazienti sottoposti a scialoadenectomia. Il tasso di successo della scialoendoscopia interventistica è di circa il 90% del totale dei pazienti trattati, come riportato in letteratura. Il tasso di recidiva è invece di circa il 10% dei casi, e il paziente dev’essere informato della possibilità di essere sottoposto ad ulteriori terapie. Un iter clinico e radiologico adeguato, rappresentato dall’ecografia, dalla TC Cone Beam 3D, dalla scialorisonanza magnetica, è indispensabile per ridurre il rischio di insuccesso e quindi di recidiva della patologia ostruttiva salivare; inoltre, in quest’ultimo caso, il chirurgo dovrebbe essere sufficientemente esperto per gestire ulteriori procedure endoscopiche e/o chirurgiche, come ad esempio l’approccio transorale/transfacciale scialoendoscopico-assistito, o procedure imaging guidate in caso di condizioni anatomiche difficoltose. La tossina botulinica e la scialoadenectomia tradizionale dovrebbero essere considerate come possibili opzioni di salvataggio in caso di fallimento di tutte le terapie conservative.
Over the last 20 years, interventional sialendoscopy has become the therapy of choice for the management of obstructive salivary disorders, favouring a significant reduction in the number of patients undergoing traditional sialadenectomy. The overall success rate of sialendoscopy is around 90% as reported by the largest case series published; recurrence is expected in about 10% of cases, and the patient should be informed about the possibility to undergo further conservative procedures to improve symptoms. Adequate pre-operative assessment, based on ultrasonography, cone beam 3D CT and MR-sialography, is mandatory to reduce the risk of unsuccessful procedures and, therefore, recurrence of obstructive sialadenitis; moreover, in case of recurrence, the surgeon should be experienced enough to manage these cases and be able to perform additional interventions, such as transoral/transfacial sialendoscopy-assisted procedures and imaging-assisted procedures in case of difficult anatomical situations. Botulinum toxin injection and traditional sialadenectomy would be considered as possible salvage treatments in case of failure of all conservative approaches.

UNASSIGNED: Sialendoscopy is a minimally invasive diagnostic and therapeutic technique used in the treatment of various salivary gland diseases. To date, there are very few suitable training models other than the pig\'s head, which has been used at the European Sialendoscopy Training Center for 22 years. The goal of this study was to describe an ovine model for sialendoscopy training and compare the ovine model\'s to the human anatomy. We propose a step-by-step approach for sialendoscopy training using this ex-vivo model.
UNASSIGNED: The anatomy of the ovine salivary ducts and glands was assessed by magnetic resonance imaging using one fresh ovine head. Thereafter, the model was designed during dissection by an experienced sialendoscopist. The various steps were then validated during consecutive dissections using a Likert-scale questionnaire.
UNASSIGNED: The full model was described in the form of a dissection guide and allowed reliable diagnostic sialendoscopy in 10/10 Stenson\'s and in 5/10 Wharton\'s ducts. Moreover, interventional sialendoscopy was simulated to provide a training model for the removal of sialoliths in the Stenson\'s duct. The human and ovine anatomy are quite similar allowing a training experience close to reality.
UNASSIGNED: We developed and evaluated an ovine model with the goal of improving training in diagnostic and interventional sialendoscopy. In particular, the Stenson\'s duct can be successfully prepared, probed and subjected to sialendoscopy. The realistic anatomical environment and excellent tissue quality created a life-like training experience for an experienced sialendoscopist. Further studies with beginners are necessary to validate this model as a training model.
UNASSIGNED: 4.

BACKGROUND: Juvenile recurrent parotitis (JRP) is characterized by recurrent episodes of painful parotid swelling in children. The purpose of this systematic review was to determine the diagnostic and therapeutic effectiveness of sialendoscopy in children affected by JRP.
METHODS: A systematic literature search was performed in PubMed, EMBASE, Scopus and the Cochrane Library until April 2022, without language restrictions or specified start date. Quality assessment was performed using the Newcastle-Ottawa Scale (NOS).
RESULTS: Our review included 524 patients and 646 sialendoscopies. The sample sizes of the different studies ranged from 3 to 77 subjects. Most authors performed sialendoscopy under general anesthesia. The mean percentage of recurrences observed was 25.1% (95% confidence intervals) (CI 23.6-26.6). There was a statistically significant relationship between the number of attacks/year and recurrences (p < 0.05). The percentage of recurrences according to the type of irrigation/flushing used ranged from 22.2% to 25.2%, with no significant differences between the use of corticosteroids alone (25.2% of recurrences), corticosteroids plus antibiotics (25% of recurrences) or saline alone (22.2% of recurrences). Sialoendoscopy has proved in all cases to be a valid method for the diagnosis of JRP, but it does not allow a reliable differential diagnosis with other autoimmune parotitis such as Sjögren\'s syndrome.
CONCLUSIONS: According to our results, parotid sialoendoscopy was 74.9% effective as a primary treatment in the prevention of recurrent symptoms in JRP. The type of ductal irrigation used did not significantly influence the prognostic outcome.

BACKGROUND: Childhood Sjögren\'s Disease (cSjD) is an underdiagnosed phenomenon with clinical and pathophysiological nuances in contrast to Sjögren\'s Disease (SjD) in the adult population. While adults typically experience sicca symptoms, children with cSjD often present with recurrent parotitis, diverse autoantibody profiles, and renal and neurological manifestations. Diagnosis and classification in pediatric rheumatology remain controversial due to the reliance on adult-focused diagnostic criteria and the lack of standardized treatment and understanding of outcomes. The purpose of the paper is to propose a multimodal treatment plan and demonstrate the effectiveness of sialendoscopy in the management of cSjD.
METHODS: We present the case of a twelve-year-old female diagnosed with cSjD using the 2016 American College of Rheumatology (ACR) and the European League Against Rheumatism (EULAR) diagnostic criteria for SjD. In addition to medical management, she underwent sialendoscopy with triamcinolone irrigation under sedation and was monitored for progress via salivary gland ultrasonography (SGUS). Over the course of one year, she demonstrated significant improvement in symptoms, with serial SGUS scores gradually decreasing by five points.
CONCLUSIONS: This paper proposes a multimodal treatment plan involving sialendoscopy and medical management as a non-invasive and potentially more effective approach for cSjD. Standardized monitoring through SGUS scoring allows objective and quantifiable measurement of treatment progress, enabling better assessment of glandular tissue status. Recurrence is possible, and each cSjD patient may present differently. Nevertheless, our year-long observation of a patient with cSjD demonstrates that sialendoscopy, as seen in adults, can promote remission of recurrent parotitis in children as well.

OBJECTIVE: Sialendoscopy is a minimally invasive procedure used to diagnose and treat obstructive salivary gland diseases. Previous studies in the topic have shown mixed results. The present study aimed to evaluate the efficacy and safety of sialendoscopy through previous systematic reviews for different outcomes of several diseases. We also aimed to assess studies\' methodological quality and heterogeneity.
METHODS: We conducted a comprehensive systematic literature search of Pubmed, Embase, Lilacs and Cochrane Library. We included systematic reviews and meta-analyses that used sialendoscopy to treat both lithiasic and alithiasic salivary glands diseases. Data extraction included studies\' characteristics and results. We assessed studies\' methodological quality using the AMSTAR-2 (A MeaSurement Tool to Assess systematic Reviews 2) tool.
RESULTS: 13 studies were included in the review, being 9 in adult populations and 4 in pediatric populations. Sialendoscopy proved to be effective at the treatment of different lithiasic and other obstructive diseases, but with important heterogeneity. The technique was also considered highly safe in most studies. However, studies had a critically low quality of evidence.
CONCLUSIONS: Most studies demonstrated high efficacy and safety of sialendoscopy, but with critically low quality of evidence. We still lack randomized studies in this field, and future systematic reviews on the topic should follow current guidelines to improve conduction and reporting.

Sjögren\'s syndrome (SS) is an autoimmune disease characterized by the involvement of exocrine glands such as the salivary and lacrimal glands. The minor salivary glands, from which tissue samples may be obtained, are important for the diagnosis, evaluation of therapeutic efficacy, and genetic analyses of SS. In the onset of SS, autoantigens derived from the salivary glands are recognized by antigen-presenting dendritic cells, leading to the activation of T and B cells, cytokine production, autoantibody production by plasma cells, the formation of ectopic germinal centers, and the destruction of salivary gland epithelial cells. A recent therapeutic approach with immune checkpoint inhibitors for malignant tumors enhances the anti-tumor activity of cytotoxic effector T cells, but also induces SS-like autoimmune disease as an adverse event. In the treatment of xerostomia, muscarinic agonists and salivary gland duct cleansing procedure, as well as sialendoscopy, are expected to ameliorate symptoms. Clinical trials on biological therapy to attenuate the hyperresponsiveness of B cells in SS patients with systemic organ involvement have progressed. The efficacy of treatment with mesenchymal stem cells and chimeric antigen receptor T cells for SS has also been investigated. In this review, we will provide an overview of the pathogenesis of salivary gland lesions and recent trends in therapeutic approaches for SS.

In sialendoscopy, ducts are dilated and the salivary glands are irrigated with saline. Contrast-enhanced ultrasound sialendoscopy (CEUSS), using microbubbles, may facilitate the monitoring of irrigation solution penetration in the ductal system and parenchyma. It is imperative to test CEUSS for its safety and feasibility in Sjögren\'s syndrome (SS) patients. CEUSS was performed on 10 SS patients. The primary outcomes were safety, determined by the occurrence of (serious) adverse events ((S)AEs), and feasibility. The secondary outcomes were unstimulated and stimulated whole saliva (UWS and SWS) flow rates, xerostomia inventory (XI), clinical oral dryness score, pain, EULAR Sjögren\'s syndrome patient reported index (ESSPRI), and gland topographical alterations. CEUSS was technically feasible in all patients. Neither SAEs nor systemic reactions related to the procedure were observed. The main AEs were postoperative pain (two patients) and swelling (two patients). Eight weeks after CEUSS, the median UWS and SWS flow had increased significantly from 0.10 to 0.22 mL/min (p = 0.028) and 0.41 to 0.61 mL/min (p = 0.047), respectively. Sixteen weeks after CEUSS, the mean XI was reduced from 45.2 to 34.2 (p = 0.02). We conclude that CEUSS is a safe and feasible treatment for SS patients. It has the potential to increase salivary secretion and reduce xerostomia, but this needs further investigation.