Although the maxillary permanent canine is frequently misplaced, misplacement of the mandibular canine is a comparatively rare event. Pre-eruptive migration of a tooth across the midline is called transmigration. This condition has not been reported for teeth other than canines. Dentigerous cysts can involve any tooth, but mandibular canines are rarely affected like premolars and molars. Odontogenic cysts associated with impacted teeth are usually removed through an intraoral approach. However, on rare occasion, an extraoral approach is required to remove these lesions. A case of transmigrant mandibular canine associated with a dentigerous cyst that required an extraoral submental approach for removal is presented. Keywords: dentigerous cyst, extraoral approach, transmigrant canine