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Congenital submandibular duct atresia (CSMDA) is a rare congenital oral lesion that appears as a cystic swelling along the course of the submandibular duct (SMD) without a lumen. Several cases of CSMDA have been reported with varying presentations and management. This review aims to identify the clinical characteristics and recovery outcomes among infants with CSMDA. The review was conducted by searching several data bases from inception to August 2023 following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. The search identified 234 studies, with 25 meeting the inclusion criteria. A total of 33 infants aged 0 days to 22 months were included, with a male predominance (75.8%). About 72.7% had unilateral cystic swelling. Infants with bilateral CSMDA (87.5%) were more likely to present with swallowing difficulties than unilateral CSMDA. Around 12% of the included children recovered spontaneously. Marsupialization of SMD is the most favored mode of treatment (83%) with no documented recurrence. Surgical treatment is recommended for infants presenting with swallowing difficulties. Yet, the quality of evidence is inadequate to recommend a widespread mode of treatment until a better quality study has been completed.
Cite this article as: Saniasiaya J, Toll E, McCaffer C, Meer GVD. Clinical characteristics and management of congenital submandibular duct atresia: A systematic review. B-ENT Published online April 16, 2025. doi:10.5152/B-ENT.2025.231518