| Summary: | Oromandibular dystonia is defined as a focal dystonia that manifests as forceful contractions of the face, jaw, and/or tongue. Lingual dystonia is a rare subtype of oromandibular dystonia that specifically affects the tongue. Multiple etiologies are thought to attribute to oromandibular dystonia, including brain damage, the use of neuroleptic medications, neurodegenerative disorders, metabolic disorders, neurodevelopmental disorders, and viral infections. Idiopathic cases of isolated lingual dystonia are rare and seldom reported in the literature. This report describes a 35-year-old female patient with lingual dystonia that was present at rest and aggravated during speech. Despite detailed history taking and a thorough examination, along with multiple imaging and laboratory studies, no cause could be established and her case was classified as being that of an idiopathic etiology.
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