Despite extensive use of antipsychotic drug treatment, few studies address the prevalence of tardive dyskinesia (TD) in homogeneous ethnic groups such as the Chinese. This study examined gender-specific relationships between TD and symptom levels in schizophrenia among Han Chinese, which have previously received little systematic study. Five hundred and twenty-two inpatients with schizophrenia receiving long-term treatment with antipsychotics were evaluated with the AIMS. The patient’s psychopathology was assessed with the Positive and Negative Syndrome Scale (PANSS). Demographic and clinical data were collected from a detailed questionnaire and medical records. The overall TD prevalence was 33.7% with rates of 39.2% (138/352) in males and 22.4% (38/170) in females (χ 2 = 14.6, df = 1, p < 0.0001; adjust odds ratio 2.06; CI, 1.32–3.16). The AIMS score in patients with TD was lower in females than males (5.3 ± 3.9 vs 6.7 ± 3.7, t = 2.52, p < 0.01) after adjustment for the significant covariates. TD was associated with the negative symptoms on the PANSS in both genders, and with age, PANSS total and positive symptoms in men, not women. Our present findings suggest that there are gender differences in the prevalence, risk, and clinical correlates of TD in schizophrenia. Although this study is limited by cross-sectional designs, the magnitude of these gender-specific differences is substantial and deservers further prospective study.