Pisa syndrome, manifested with persistent lateral flexion of the trunk, is most commonly associated with prolonged treatment with typical antipsychotics. However, it was also reported as occurring with atypical antipsychotics. To our knowledge, there have been very few reports of clozapine-associated Pisa syndrome. Here we report 1 case of Pisa syndrome in a 39-year-old woman with schizoaffective disorder who developed tonic flexion of trunk and head toward the left side after clozapine treatment (400 mg/d) for 5 months. Clozapine was reduced to 25 mg/d within 15 days; the dystonic reaction then completely resolved within the next 3 to 4 weeks. Caution should be taken while prolonged use of clozapine in patients with risk factors of Pisa syndrome.