"Ms. M," a 42-year-old woman, was admitted with treatment-refractory schizophrenia. She was given full dose of aripiprazole, paliperidone, quetiapine, and amisulpiride, as well as electroconvulsive therapy (ECT), with limited improvement. Because of refractory psychotic symptoms, clozapine was gradually increased from 25 mg/day to 375 mg/day within 1½ months. However, her physical examination revealed tonic flexion of the neck to the right, with medial rotation. She had no other neurological deficits and no history of dystonia. The angle between the head and spine increased to 60° in the next 3 weeks. Ms. M received biperiden 8 mg/day and clonazepam 4 mg/day while the clozapine dose was tapered to 250 mg/day. However, her torticollis persisted, and psychotic symptoms were aggravated. We added aripiprazole 15 mg/day for 2 weeks, and the torticollis partially improved. It did not worsen when we increased clozapine to 325 mg/day in the next 2 weeks. The bending angle decreased from 15° to 0° when we increased aripiprazole to 30 mg/day for another 4 weeks. Her psychotic symptoms were also better controlled.