Primary extracranial meningiomas are uncommon; they have been documented in the ear, skin, orbit, parapharyngeal space, parotid gland, mediastinum, and rarely the paranasal sinuses. Tuberous sclerosis is an autosomal dominant neurocutaneous syndrome characterized by multiple connective tissue tumours. We report the first case of a tuberous sclerosis patient with a maxillary antral meningioma.