A rare case of isolated myoclonus in an elderly male without a history of epilepsy.

AIM: Through this case report we attempt to highlight the presentation, initial investigation and management of lingual myoclonus as well as consolidate relevant literature.

CASE: We present a unique case of a 72-year-old man who was admitted to the hospital for a sudden onset episodic speech arrest. Lingual myoclonus, an isolated movement disorder, manifested as an intermittent expressive aphasia secondary to the intrusion-protrusion movements of his tongue. During this time, the patient remained conscious and was able to continue to follow commands. Initial diagnostic evaluation with a CT scan, MRI and EEG failed to illicit a clear underlying etiology and the patient was empirically treated with valproic acid with complete resolution of his symptoms.

DISCUSSION: This unusual presentation represents a rare disorder which is not well described in literature. Initial evaluation of which required excluding associated etiologies including strokes, seizures, medications/toxins or CNS infections. Without a clear etiology on initial diagnostic evaluation, the patient was empirically treated as no clear guidelines exist. This case presentation is an attempt to add to the current understanding of lingual myoclonus.