We report on a 42-year-old man with paroxysmal kinesigenic dyskinesia who was referred as a refractory case to any drug used in the past as monotherapy or in combination. Our decision to discontinue his current combined medication and to administer only high-dose phenytoin led to significant improvement. It is of interest to note that the previous use of phenytoin in combination with other antiepileptic and neuroleptic drugs had no effect. In addition, the co-administration of gabapentin led to a dramatic recurrence of the episodes.