Clozapine is recommended for patients with schizophrenia and tardive dystonia (TD); however, the appropriate dose remains unclear. In this case, a low dose (150 mg/day) of clozapine improved refractory TD and further ameliorated psychiatric symptoms. Herein, we report on a 41-year-old female with schizophrenia and TD who was treated with a low clozapine dose. After eight weeks of continuous clozapine at 150 mg/day (16 weeks after clozapine initiation), her TD dramatically improved, and her psychiatric symptoms were relieved. Low clozapine doses could ameliorate refractory TD. However, this effect might require up to several weeks. Clinicians should be patient unless they consider it better to increase the clozapine dose.