A 68-year-old woman, presented with a squamous cell carcinoma of the malar region, and underwent wide local excision. During her clinical examination, repetitive protrusion and intrusion of the tongue as well as stereotypic, abnormal movements of the mouth and lips were observed, in a pattern that resembled chewing, sucking or lip pursing; dyskinesias ceased when she was speaking or bringing food to the mouth. She was unaware of the movements and the tongue was observed to move similar to choreiform movements, while revealing a giant "snake-like" macroglossia. She had history of mental retardation and alcohol abuse, and was under classic antipsychotic medications for several years. During a previous neurological investigation, type I Chiari malformation was diagnosed. In this case, concomitant Chiari malformation and neuroleptic-induced tardive dyskinesia, may together have been responsible for giant macroglossia, and to our best knowledege no similar observation has been reported in the literature. Address correspondence and reprint requests to Ricardo Horta, MD, PhD, Avenida Menéres, no 234, Bloco 2, 4° Frente Esquerdo, 4450-189 Matosinhos Sul-Porto, Portugal; E-mail: ricardojmhorta@gmail.com Received 30 June, 2018 Accepted 14 August, 2018 The authors report no conflicts of interest. Supplemental digital contents are available for this article. Direct URL citations appear in the printed text and are provided in the HTML and PDF versions of this article on the journal's Web site (http://bit.ly/2iuFjMi). © 2019 by Mutaz B. Habal, MD.
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