Salivary Cutaneous Fistula Caused by Submandibular Sialolithiasis in an Adolescent

Abstract

Pediatric submandibular sialolithiasis with a salivary cutaneous fistula is exceptionally rare. A 15-year-old boy presented with a 4-month history of a persistent right submandibular inflammatory lesion with a central opening and hypergranulation despite previous incision and drainage. Contrast-enhanced computed tomography demonstrated a 10.8 mm distal Wharton duct stone, a 34 mm inflammatory lesion extending toward the skin, and marked atrophy of the right submandibular gland. Because of persistent inflammation, fistula formation, and severe glandular atrophy, the fistulous tract and affected gland were excised. Histopathology demonstrated chronic ulceration with acute and chronic inflammation; an excised lymph node showed reactive hyperplasia. At 4 months, the fistula had resolved without recurrent inflammation. Although gland-preserving treatment should be considered in children, gland excision may be appropriate in advanced disease with marked glandular atrophy and persistent fistula formation.