We report a case of laryngeal dyskinesia resulting in severe rest and exercise dyspnea. A 51-year-old man treated for 2 years with flupentixol, an incisive neuroleptic, developed severe dyspnea due to intermittent, rhythmic, and dystonic movements of the vocal cords and upper airway. Local injections of botulinum toxin resulted in spectacular regression of laryngeal spasms and major improvement in breathing. This case emphasizes the risk of upper respiratory dyskinesias associated with neuroleptic treatment and shows the feasibility of a new local treatment in this life-threatening disorder.