Ectopic lingual thyroid is a rare developmental anomaly, presenting in the adult with obstructive symptoms during respiration, deglutition and speech. The main therapeutic issue after exact diagnosis is the restoration of thyroid function, and surgical intervention, when necessary, has to deal with problems related to the site and function of the ectopic gland. A case of a 25-year-old female with dyspnoea and dysphagia due to a 3 x 4 cm ectopic lingual thyroid, excised by an original peroral approach via a mandibular midline osteotomy, is reported. Our surgical technique is described and the diagnostic and therapeutic problems are reviewed.