Tardive dyskinesia (TD) is a movement disorder that presents with abnormal, involuntary, and irregular choreoathetoid movements involving mouth, tongue, face, trunk, or extremities after prolonged use of dopamine receptor–blocking agents. Currently, there is still a lack of effective treatment for it. Here, we report a female patient with paranoid schizophrenia, who has had persistent TD for 8 years. After electroconvulsive therapy (ECT) was performed, both of her psychotic symptoms and dyskinetic movements were improved. The efficacy and potential clinical variables of ECT in the treatment of TD were reviewed.