Macroglossia is a morphological and volumetric alteration of the tongue, caused by muscular hypertrophy, vascular malformation, metabolic diseases, and idiopathic causes and also associated with Down and Beckwith-Wiedemann syndromes. This alteration can cause dental-muscle-skeletal deformities, orthodontic instability, masticatory problems, and alterations in the taste and speech. In this paper we present a case of true macroglossia diagnosed in a female patient, 26 years, melanoderma, no family history of disease, with a history of relapse of orthodontic treatment for correction of open bite, loss of the lower central incisors, and complaint of difficulty in phonation. The patient was submitted to glossectomy under general anesthesia using the “keyhole” technique, with objective to provide reduction of the lingual length and width. The patient developed with good repair, without taste and motor alterations and discrete paresthesia at the apex of the tongue.
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