Isolated involuntary tongue movements are rare and poorly understood. The anatomical substrate and pathogenesis underlying involuntary tongue movements remain elusive. We describe a patient who developed isolated continuous rhythmic involuntary tongue movements after pontine infarct without evidence of hypertrophy of inferior olivary nucleus on follow-up magnetic resonance image. We discuss the rhythmic involuntary tongue movements as a prototype of involuntary hyperkinetic movement released by a central pacemaker.