Giant Submandibular Sialolith Presenting with Sialocutaneous and Sialo-Oral Fistula: A Case Report and Review of Literature

Abstract

Sialolithiasis is a common disease of the salivary glands and a major cause of salivary gland dysfunction. It commonly affects middle-aged and has male predominance. Submandibular gland or its duct is most commonly affected. The size of salivary calculi may vary from less than 1 mm to a few cm in the largest diameter. Salivary stones that exceed 15 mm in any dimension are classified as giant. Association of sialocutaneous or sialo-oral fistula with salivary stones is considered rare. Long-standing stones with chronic inflammatory process may lead to fistula formation.

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“…27 Sialo-oro-cutaneous fistula. 18,22 Wakoh et al presented the case of cutaneous fistula related with ectopic submandibular gland sialolith. 7 Ha et al (2020) exhibited a sialo-cutaneous fistula but with no evidence of submandibular gland sialolith.…”

“…Despite the fact that in the report of Chandak et al (2012) the case with diagnosis of acute submandibular sialadenitis complicated with abscess was established there was no radiological evidence of sialoliths in submandibular gland. 28 In the reported cases, some authors combined multiple diagnostic tools, like Jayachandran et al (X-ray, MSCT, MRI), 18 others used a single one, like Hoffman (MSCT) 8 . Among 24 complication cases presented in the Table 1, computed tomography (non-contrast/contrast enhanced) was used in 10 cases, OPG -in three cases, X-ray -in five cases, X-ray fistulography -in three cases, fluoroscopyin one case, US -in four cases, MRI -in one case, sialoendoscopy -in one case, and X-ray sialography -in one case.…”

Chronic sialadenitis with sialolithiasis associated with parapharyngeal fistula and tonsillolith

“…of patientsNo. of stonesAge, ySialolith dimensions, cmGreatest dimension, cmFistulaImaging modalityJayachandran [15]2011Case report11522.6 × 2.1 × 2.02.6Sialo-oro-cutaneousRadiography, CT, MRIDrage [9]2004Case series3352.322.0SialocutaneousCase 1—CT, Case 2—radiography, Case 3—sialogramKurtoglu [16]2015Case report11522.5 × 0.8 × 0.62.5Sialo-oralNone describedRauso [17]2012Case report11565.65.6Sialo-oralNone describedSingh [18]2015Case report11531.0 × 1.01.0SialocutaneousFistulography, CTRangappa [19]2014Case report11551.0 × 1.01.0SialocutaneousUltrasound, radiographySutay [20]2003Case report11223.7 × 0.73.7Sialo-oralRadiographyAlmasri [21]2005Case report11702.3 × 1.72.3SialocutaneousRadiography, CTKasat [6]2012...…”