A hiccup is a myoclonic jerk of the diaphragm, and cases of hiccups may last for more than 48 hours (persistent hiccups) or even more than 2 months (intractable hiccups). Current pharmacologic treatment of persistent or intractable hiccups mainly includes antidopaminergic drugs. We describe the case of a 60-year-old man with a recent diagnosis of right insular ischemic stroke who presented with frequent, intense, and disabling hiccups for more than 1 month. As diagnosis of poststroke hiccups was assumed, the patient was treated over the next 6 months with adequate doses of various antipsychotic drugs commonly used for the treatment of hiccups; however, all were discontinued because of adverse effects. Indeed, dyskinesia after chlorpromazine (up to 75 mg/day for 4 wks), as well as somnolence and dyskinesia after haloperidol (up to 6 mg/day for 6 wks), somnolence after gabapentin (up to 1800 mg/day for 8 wks), and severe somnolence and hypotension after baclofen (up to 50 mg/day for 6 wks) were reported. The patient was then prescribed tetrabenazine at a starting dose of 12.5 mg twice/day (25 mg/day), with a nearly complete remission of the hiccup symptomatology after ~6 weeks, when a daily dose of 150 mg was reached. We therefore hypothesize that a supratentorial lesion may disrupt the modulation of dopaminergic pathways involved in the regulation of medullar centers responsible for the hiccup reflex. To our knowledge, this is the first case report of poststroke hiccups responding to tetrabenazine. The dramatic response of our patient to tetrabenazine monotherapy suggests that this drug may be a valuable pharmacologic alternative for patients with hiccups after stroke who are intolerant or unresponsive to classic antipsychotic agents.