Cisapride is a widely used drug which stimulates gastrointestinal motility. We report a case of extrapyramidal syndrome apparently caused by cisapride. In November, 1993, a 77-year-old female patient had symptoms of tremor, stiffness and numbness. She had been receiving 15 mg/day of cisapride since June 1993. Additionally, the patient was receiving 10 kinds of medications.Cisapride was discontinued in December 1994. Two months later, the above-noted symptoms disappeared. Cisapride acts selectively on the myenteric plexus of the digestive tract, and enhances the release of acetylcholine. It has been reported that cisapride has neither an antidopaminergic nor a nonspecific cholinergic property. For these reason, it is believed that cisapride did not induce these side effects. However, the above case of extrapyramidal syndrome apparently caused by cisapride shows that cisapride should be used carefully especially with regards to the potential neurological side effects.