Osseous choristoma is a rare, benign lesion of the oral cavity and is usually found in the tongue. It presents as a tumour-like mass of normal bony structure with mature cells in an abnormal position. The object of this paper is to report one case of osseous choristoma.A 32-year-old male presented with a lesion in the submandibular region, which was treated by excision. After 28 months of follow-up there was no recurrence.Upon reviewing the English literature, no previous case of an osseous choristoma located in the submandibular region has been found. Extended clinical and radiographic follow-up is necessary after surgical excision of an osseous choristoma, despite its benign nature.