Sialendoscopy for recurrent sialadenitis: a 9-year retrospective review at a tertiary paediatric centre

Introduction

Sialadenitis is a frequent clinical presentation in the paediatric population with an overall unknown incidence (1). Paediatric sialadenitis is distinct from sialadenitis affecting adults, with paediatric cases commonly being either viral or idiopathic in nature (2). While most episodes are self-limiting, recurrent sialadenitis raises concern for possible underlying duct obstruction, juvenile recurrent parotitis (JRP), or paediatric onset Sjögren’s syndrome. Obstructive causes of recurrent sialadenitis include sialoliths and duct stenosis. Sialoliths most commonly affect the submandibular gland, while stenoses can be present along any of the ducts and can occur after the passage of a sialolith or may develop as a post-inflammatory complication (3).

JRP is characterised by recurrent, painful swelling affecting either or both of the parotid glands, with submandibular gland involvement seen less commonly (4). Management of JRP flares typically includes the use of non-steroidal anti-inflammatory agents, supportive care, and antibiotics when simultaneous bacterial infection is suspected. JRP is considered a condition of early childhood, and most individuals will outgrow the condition during the second decade of life (4). An estimated 1% of all adult patients with Sjögren’s syndrome will have symptom onset evident in childhood, characterised by recurrent sialadenitis (5).

Sialendoscopy is a minimally invasive technique, allowing visualisation and treatment of underlying ductal pathology. Sialendoscopy is performed by inserting a fine semi-rigid endoscope into the duct draining a major salivary gland and is implemented in the paediatric setting under anaesthesia. Possible causative findings include duct stenosis and sialoliths. In cases of general inflammatory change, characteristic findings include pale ductal mucosa and sludge or debris within the ductal lumen. When one of these anomalies is identified, the operator may intervene via working channels within the endoscope to remove stones, flush intraductal debris, and dilate stenotic regions by use of catheter balloons or stent placement. In cases of inflammatory sialadenitis, as is seen in JRP and Sjögren’s syndrome, sialendoscopy offers an avenue for therapeutic intervention whereby irrigation of salivary ducts with saline and topical steroids has been shown to reduce the frequency and severity of salivary gland inflammation (6).

This study adds to the limited literature by reporting on one of the larger Australian paediatric cohorts who have undergone sialendoscopy.

The primary aim was to evaluate the efficacy of sialendoscopy in managing recurrent paediatric sialadenitis, measured by patient-reported changes in the frequency and severity of sialadenitis episodes following intervention. Outcomes were analysed on a per-procedure basis. Secondary objectives included characterising intra-operative findings, stratifying outcomes based on indication for sialendoscopy, as well as reporting associated complications and describing our institutional technique.

Methods

A retrospective analysis was undertaken, reviewing cases of sialendoscopy performed between December 2015 and December 2024 by the Department of Paediatric Otolaryngology Head and Neck Surgery at The Children’s Hospital at Westmead in NSW, Australia. Data gathered included patient demographics, indication for surgery, pre-operative imaging findings, surgical technique, intra-operative findings, surgical complications, and recurrence of sialadenitis reported on clinical follow-up. Families were encouraged to keep a personal diary or record of frequency and severity of episodes of sialadenitis; however, no standardised or validated outcome questionnaire was used. Patients who reported a reduction in the severity or frequency of episodes on clinical follow-up were considered to have improved clinically. Patients who reported a similar occurrence of episodes of sialadenitis were considered to have had no improvement postoperatively. The study was conducted in accordance with the Declaration of Helsinki and its subsequent amendments. Ethics approval was granted from the Sydney Children’s Hospital Ethics Committee (No. 2024/ETH01977). A waiver of consent was approved for the retrospective analysis and pooled, non-identifiable analysis of healthcare data. This study is reported according to the STROBE reporting guidelines (available at https://www.theajo.com/article/view/10.21037/ajo-25-68/rc).

Criteria for inclusion were: sialendoscopy performed for all indications on paediatric patients with accessible operation reports and medical record data, including patients who had already undergone previous sialendoscopy, with overall results analysed on a per-procedure basis. Indications for sialendoscopy included JRP, sialoliths, duct stenosis, and Sjogren’s disease. JRP was defined in this study as recurrent inflammatory parotitis without an overt underlying cause. Exclusion criteria included patients with insufficient procedural or clinical follow-up data, as well as patients booked for sialendoscopy but in whom sialendoscopy could not be performed. Cases where sialendoscopy could not be performed were due to the inability to cannulate the duct or advance instruments within the duct. Missing data were handled using complete-case analysis only, with no statistical imputation performed.

All cases were performed by a single surgeon under general anaesthesia with the use of a bite block and initial use of lacrimal probes to cannulate and dilate ducts. In cases of sialendoscopy of the submandibular gland, retro-papilla ductotomy was performed when it was not possible to cannulate the ducts prior to sialendoscopy. Examination was then performed with a three-channel, 1.3 mm endoscope (Karl Storz). Prior to completion of sialendoscopy, patients with a background of inflammatory sialadenitis would routinely receive triamcinolone acetonide 40 mg/mL (Kenacort^®-A 40) administered via irrigation from the endoscope with 1 mL of volume administered. When performed as an independent procedure, all patients were planned for discharge the same day of the procedure.

Statistical analysis

Descriptive statistics were used to summarise patient demographics, procedural characteristics, intra-operative findings, and outcomes. Categorical variables are reported as frequencies and percentages. Comparisons of reported clinical improvement across indications for sialendoscopy were performed using Fisher’s exact test. This test was selected due to small sample sizes within several indication subgroups with limited power, and the presence of expected cell counts less than five, for which Chi-squared testing would be inappropriate. Statistical significance was defined as P<0.05. Analyses were performed on a per-procedure basis using available complete cases only.

Results

Between December 2015 and December 2024, 38 patients underwent sialendoscopy with 46 total procedures performed. One patient was excluded due to the inability to cannulate the duct at the time of surgery. Follow-up data were unavailable for 2 patients (4.4%) and were due to non-attendance, yielding a final cohort of 35 patients with 43 procedures for analysis. Patient demographics and procedural characteristics are summarised in Table 1.

Patients were offered repeat sialendoscopy in cases where there had been a clinically noticeable reduction in the severity and frequency of episodes of inflammatory sialadenitis, but had then begun to experience a recurrence of episodes in the months or years following. Sialendoscopy was performed twice in six patients, while one patient underwent sialendoscopy three times across 3 years. Age at time of procedure ranged from 19 months to 17 years (mean, 9.7 years; standard deviation, 4.0 years). Of all procedures, 56% (n=24) were performed on female patients.

Five patients remained admitted overnight, whilst all others went home on the day of surgery. Follow-up duration was heterogeneous, with a median of 179 days (interquartile range, 30–503 days) and a mean follow-up of 354±457 days.

Indications for sialendoscopy were primarily recurrent sialadenitis, with patients meeting diagnostic criteria for JRP (n=27). Other indications included recurrent sialadenitis with imaging suggestive of a stone (n=9) or a suspected ductal stricture (n=3). Sialendoscopy was performed for 2 patients with either a known background or subsequent diagnosis of Sjögren’s syndrome, and one of these patients underwent sialendoscopy three times. Indications for sialendoscopy and associated intra-operative findings are detailed in Table 2.

No major procedure-related complications were observed. Minor adverse events occurred infrequently and included inability to retrieve a stone (n=3), minor ductal injury (n=2), and epistaxis related to nasal intubation (n=1).

Outcomes were analysed on a per-procedure basis. Overall, patient-reported clinical improvement was observed following 39 of 43 procedures (91%). When stratified by indication, improvement was reported in 25 of 27 procedures performed for JRP (93%), 7 of 9 procedures for sialolithiasis (78%), 3 of 3 procedures for suspected ductal stricture (100%), and 4 of 4 procedures for Sjögren’s syndrome (100%).

Comparisons of reported improvement across indications did not demonstrate statistically significant differences (all P>0.05). Given the small sample sizes within individual indication groups, particularly for suspected ductal stricture and Sjögren’s syndrome, these analyses were interpreted descriptively rather than as evidence of equivalence or superiority (Table 3). Of patients who reported minimal therapeutic benefit from sialendoscopy, one underwent further investigation and was thereafter found to have chronic non-bacterial osteomyelitis of the mandible, which was managed with bisphosphonates. Three patients with sialoliths had stones that were unable to be retrieved at the time of sialendoscopy. On follow-up, one of these patients had no further episodes of sialadenitis and so was not booked for further surgery. Another patient had ongoing recurrent sialadenitis and so was booked for repeat sialendoscopy, with subsequent successful endoscopic removal of the stone. One patient was booked for repeat sialendoscopy, with an unsuccessful repeat attempt at stone removal, prompting removal of the submandibular gland during the same anaesthetic. While both patients with paediatric Sjögren’s syndrome did report an initial reduction in severity and frequency of episodes of sialadenitis, they both continued to experience recurrent mild episodes with a return of usual frequency within 1–2 years after sialendoscopy.

Discussion

This study demonstrates that sialendoscopy is a safe intervention associated with high rates of patient-reported clinical improvement in the paediatric setting for recurrent sialadenitis. Our findings are consistent with published literature reporting favourable outcomes (7,8). Interpretation of efficacy in cases of JRP must be weighed against the natural history of the disease. JRP is a self-limiting inflammatory condition of early childhood, characterised by recurrent episodes of sialadenitis that typically decrease in frequency and severity with age (9). Spontaneous remission is most commonly observed during the second decade of life (10). This high rate of natural resolution presents an inherent challenge when evaluating the true impact of therapeutic interventions. Improvements observed following sialendoscopy can partially reflect the expected natural course of JRP rather than a direct procedural outcome. This limitation is emphasised when performing retrospective analysis without a control group or standardised outcome assessment.

Despite this, several factors suggest that sialendoscopy may still play a clinically meaningful role in selected patients with JRP. Many children included in this cohort experienced frequent or severe episodes prompting referral to a tertiary centre, suggesting a subgroup with more burdensome disease. The temporal association between intervention and reported symptomatic improvement, as well as the recurrence of symptoms in some patients that responded again to repeat sialendoscopy, supports a therapeutic effect beyond spontaneous remission alone. Furthermore, intra-operative findings of ductal debris, pale mucosa, and inflammatory changes lend biological plausibility to the observed benefit of ductal irrigation and topical steroid administration in reducing inflammatory burden.

In contrast to JRP, the role of sialendoscopy in obstructive sialadenitis is more clearly defined. In this cohort, patients with ductal strictures or sialolithiasis generally reported improvement when the obstructive pathology could be successfully addressed endoscopically, consistent with established adult and paediatric literature (11,12). These findings reinforce sialendoscopy as a minimally invasive alternative to open surgery in appropriately selected paediatric patients with obstructive salivary gland disease.

Patients with paediatric-onset Sjögren’s syndrome represented a small subset of this cohort, limiting firm conclusions. While transient symptomatic improvement was reported, ongoing mild episodes of sialadenitis persisted, reflecting the chronic autoimmune nature of the condition. In this context, sialendoscopy may serve as an adjunctive symptomatic treatment rather than a definitive therapy (13).

Several limitations warrant consideration. The retrospective design and reliance on patient-reported outcomes without a validated scoring system necessitated a binary classification of clinical response, limiting precision in analysis. While no validated questionnaires currently exist to assess the severity of JRP, there may be a role for adapting symptom-based instruments developed for adult populations (14).

Follow-up duration was heterogeneous, and a small number of patients were lost to follow-up, largely due to the tertiary referral nature of the service. Most importantly, the absence of a control group precludes definitive attribution of symptom improvement to sialendoscopy, particularly in conditions with a known tendency toward spontaneous resolution, such as JRP.

Conclusions

Overall, while recognising the natural history of JRP and its propensity for spontaneous remission, the findings of this study support sialendoscopy as a safe, low-morbidity intervention that may provide symptomatic benefit in select paediatric patients with recurrent sialadenitis. Prospective studies incorporating standardised outcome measures and longer-term follow-up are required to more accurately delineate the magnitude and durability of benefit attributable to sialendoscopy and to better define its role within the management algorithm of JRP.

Acknowledgments

None.

Footnote

Reporting Checklist: The authors have completed the STROBE reporting checklist. Available at https://www.theajo.com/article/view/10.21037/ajo-25-68/rc

Data Sharing Statement: Available at https://www.theajo.com/article/view/10.21037/ajo-25-68/dss

Peer Review File: Available at https://www.theajo.com/article/view/10.21037/ajo-25-68/prf

Funding: None.

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://www.theajo.com/article/view/10.21037/ajo-25-68/coif). The authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. The study was conducted in accordance with the Declaration of Helsinki and its subsequent amendments. Ethics approval was granted from the Sydney Children’s Hospital Ethics Committee (No. 2024/ETH01977). A waiver of consent was approved for the retrospective analysis and pooled, non-identifiable analysis of healthcare data.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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