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Submandibular lithiasis: the risk of neoplasia masked by the stone

Submandibular sialolithiasis represents the most common cause of salivary obstruction, ma...

Clinical context and regional challenges

Submandibular sialolithiasis represents the most frequent cause of salivary obstruction, but its management in the Drâa-Tafilalet region (Morocco) faces specific geographical and environmental constraints. In this rural area, exposure to extreme heat (>45°C), consumption of mineral-rich well water, and limited access to specialized care promote dehydration and stone formation. Contrary to current standards favoring minimally invasive techniques such as sialendoscopy, these conditions lead to major consultation delays. Practitioners then face advanced chronic sialadenitis, irreversible glandular fibrosis, or recurrent suppurations, often making submandibulectomy inevitable. A crucial diagnostic risk persists: the presence of a stone can mask an underlying glandular neoplasia, undetectable clinically or by conventional imaging.

Study objective and hypotheses

This retrospective descriptive study, conducted on 23 patients operated on at Al Amir Sultan Hospital between January 2024 and December 2025, aims to analyze the epidemiological, clinical, and radiological characteristics of patients undergoing glandular excision in this specific context. The primary objective is to evaluate the contribution of systematic histopathological examination of resection specimens. The authors test the hypothesis that regional risk factors and the delay in management increase not only glandular morbidity but also the probability of discovering associated malignant pathologies, masked by the initial lithiasic symptomatology.

Methodology: a retrospective study in a rural setting

This retrospective descriptive study, conducted between January 1, 2024, and December 31, 2025, at the Al Amir Sultan Hospital Center in Errachidia (Morocco), included 23 patients who underwent submandibulectomy for calculous sialadenitis. Cases of anterior ductal stones treated by simple transoral extraction, autoimmune or radiation-induced sialadenitis, as well as neoplasms diagnosed preoperatively, were excluded.

The evaluation protocol systematically included:

  • Preoperative assessment: Clinical history, routine biology, dental panoramic radiograph and cervical ultrasound for the entire cohort (n=23).
  • Additional imaging: A computed tomography (CT) scan was performed in 8 patients (34.8%) in cases of suspected deep extension or trismus.
  • Surgical technique: Standard cervical approach under general anesthesia, with preservation of the marginal mandibular branch of the facial nerve and proximal ligation of Wharton's duct.
  • Histopathological analysis: Systematic examination of excision specimens (10% formalin fixation, hematoxylin and eosin staining).

Post-operative follow-up was rigorously documented at one week, one month, three months, and six months. The descriptive analysis, performed via Microsoft Excel 2019, also integrated specific regional variables: thermal exposure (> 45°C), consumption of highly mineralised well water, and average distance from home to the reference centre (142 ± 67 km).

Epidemiological and clinical profile: the impact of delayed management

The study included 23 patients (13 men and 10 women, ratio 1.3:1) with a mean age of 51.1 ± 9.8 years. The geographical context of the Drâa-Tafilalet region weighs heavily on the clinical picture: 78.3% of patients reside in rural areas more than 80 km from the hospital center (average of 142 ± 67 km). This isolation, coupled with a complex care pathway, results in an average consultation delay of 24.2 ± 8 months after the onset of the first symptoms.

At the time of management, 95.7% of patients suffered from submandibular swelling triggered by meals, and 60.9% had a history of purulent episodes or abscesses requiring drainage. Before their transfer, 73.9% of patients had already received at least two courses of antibiotic therapy for recurrent infections.

Diagnostic and operative data

Cervical ultrasound, performed systematically, identified calcifications in 100% of cases. Computed tomography (CT) was used in 34.8% of patients (n=8) to evaluate deep extensions or cases of trismus. Calculus dimensions ranged from 5 to 19 mm (mean 8.9 ± 2.7 mm).

Clinical / operative parameterAverage value (n=23)
Duration of the procedure44 ± 12.1 minutes
Length of stay2.2 days
Average calculus size8.9 mm
Post-operative complication rates8.7% (n=2)

Sur le plan chirurgical, aucune complication per-opératoire n'a été relevée. En post-opératoire, deux complications mineures (8,7 %) ont été notées : une parésie transitoire du rameau marginal du nerf facial (résolue à 3 mois) et une infection superficielle de la plaie.

Histopathology: the incidental discovery of malignancy

The systematic histopathological examination of the excision specimens confirmed chronic sclerosing sialadenitis with fibrosis or parenchymal atrophy in 21 patients (91.3%). However, one result deserves particular attention: in two patients (8.7%), histology revealed unsuspected malignant neoplasms, clinically masked by lithiasic symptomatology:

  • Low-grade mucoepidermoid carcinoma: 2.2 cm tumour, classified pT2N0M0.
  • Adenoid cystic carcinoma: 2.1 cm tumour, classified pT2N0M0, with presence of perineural invasions.

These two cases concerned elderly men (63 and 71 years old) presenting with progressive glandular induration. Although lithiasis was confirmed by imaging, it masked an underlying tumour process that required, after surgery, adjuvant radiotherapy.

The lithiasic tree that hides the tumoral forest

This retrospective study conducted on 23 patients in the Drâa-Tafilalet region highlights a brutal clinical reality: diagnostic delay transforms a common obstructive pathology into an irreversible fibrous condition. With an average consultation delay of 24.2 months, submandibulectomy is required not as a first-line therapeutic choice, but as the sole recourse against chronic sclerosing sialadenitis (91.3% of cases). Geographical distance (142 km on average) and extreme climatic conditions promoting dehydration are major determinants here of the severity of the clinical presentations observed.

The most striking result remains the incidental discovery of two carcinomas (mucoepidermoid and adenoid cystic) within glands clinically and radiologically labeled as lithiasic. This 8.7% rate of associated malignancy serves as a reminder that the presence of a calculus (ranging from 5 to 19 mm in this series) can mask an underlying neoplastic process, particularly in elderly patients presenting with progressive glandular induration.

The study presents methodological limitations inherent to its clinical context: the modest sample size (n=23) and the absence of advanced imaging (MRI) or fine-needle aspiration cytology (FNAC) limit preoperative characterization. However, the results confirm the safety of the standard cervical approach with a mean operative time of 44 minutes and a low rate of nerve morbidity (4.3% transient marginal nerve paresis).

In practical terms, for the practitioner:

  • Suspicion of malignancy: Do not consider the presence of a calculus as an exclusion diagnosis; marked induration or constitutional signs require increased vigilance, as lithiasis can mask an underlying tumor process (8.7% of cases here).
  • Systematic histopathology: Histological examination of any excised submandibular gland is a medico-legal and clinical obligation, regardless of the macroscopic appearance or preoperative imaging results.
  • Environmental factors: In regions with high heat or highly mineralized water, strengthen patient education on hydration to prevent salivary stasis and lithogenesis.

Technical lexicon

Sialolithiasis: A condition characterized by the formation of salivary stones, constituting the most common cause of glandular obstruction (approximately 50% of major salivary gland pathologies).

Sialendoscopy: Minimally invasive diagnostic and treatment procedure allowing for gland preservation, used for endoluminal exploration of the ductal system.

Submandibulectomy: Surgical procedure consisting of the total excision of the submandibular gland, performed here via a standard cervical approach in cases of irreversible lesions or suspected tumour.

Wharton's duct: Excretory duct of the submandibular gland; its proximal ligation is a crucial step during the surgical excision of the gland.

Glandular neoplasm: Underlying salivary tumor whose presence may be clinically masked by lithiasis and which requires systematic histopathological examination.

Fibrosis: Irreversible fibrous transformation of the glandular parenchyma secondary to chronic obstructive sialadenitis or recurrent episodes of suppuration.


Source

  • Original title: Submandibulectomy for Lithiasic Submandibular Sialadenitis: A Case Series of 23 Patients in the Drâa-Tafilalet Region of Southeastern Morocco
  • Authors: Abdelouahid Taleuan, Mohammed ElAmine Squalli Houssaini, Adnane Lakhal, Abdellatif Oudidi
  • Publication: Cureus - 2026-07-12
  • DOI: https://doi.org/10.7759/cureus.112555

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