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Abstract Congenital agenesis of the submandibular gland with compensatory hypertrophy of the contralateral gland is an extremely rare disorder and only few cases are reported in the literature. To our knowledge the reported cases of submandibular agenesis have been asymptomatic and associated with facial anomalies. In our study we present the imaging features of the right submandibular gland agenesis with associated capillary hemangioma of the cartilaginous nasal septum in a middle-aged man who presented with a right-sided nasal block.