Dermoid cysts into ENT zone are specially infrequent--just the 7% of the organism--, and extremely rare if are located in the tongue. They are usually detected as midline acute swelling, and produce swallowing, speech and sleep disorders at neonatal period because of their congenital character. We present an eleven-year-old boy admitted at our Hospital bearing a submandibular and oral swelling, and dysphagia with odynophagia, finally diagnosed as an intralingual double dermoid cyst of the anterior two thirds. The history of frenulectomy due to a presumed ankyloglossia, the previous presence of a dorsal tumour in tongue with a difficult phonetic articulation, and the midline location of lesions made us to suspect on dermoid cysts of the tongue. Complete surgical excision were diagnostic and therapeutic, and the child recovered totally their mastication, swallowing and speech abilities. Pathophysiological aspects, treatment and the atypical course of the case are discussed.