Canalicular adenoma (CA) is a rare, unique benign salivary gland neoplasm, which usually involves the minor salivary glands of the upper lip, buccal mucosa and palate. It is usually seen in middle-aged or older individuals, has a female predilection, and commonly presents as a painless, slow-growing, non-ulcerated nodule or swelling. Its treatment involves surgical excision or enucleation. This case report describes a case of CA of the ventrum of the tongue diagnosed in a 41-year-old male patient, which could possibly be the first reported case of this lesion involving the tongue.