To elucidate a widely suspected but inconclusive association between rs6280 in the dopamine receptor 3 gene (DRD3) and prevalence of tardive dyskinesia (TD), we conducted a meta-analysis of studies obtained in a systematic search of several bibliographic systems. We conducted several analyses of funnel plot asymmetry, overall heterogeneity, and study characteristics in analyses analogous to general, dominant and recessive inheritance models with the prevalence odds ratio (POR) as the measure of association. Thirteen eligible studies were identified with publication dates between 1997 and 2008. Evidence of funnel plot asymmetry was discerned in the dominant and general model analyses, but not in the recessive model analysis. Stratified analyses indicated that publication year, TD assessment method (Schooler-Kane criteria or other) and TD assessment frequency (single or repeated) were important study characteristics associated with heterogeneous PORs across studies. Studies conducted among patients with older age, fewer women or European (compared with Asian) ancestry reported stronger average PORs. Summary POR estimates under the dominant and general inheritance models were not warranted due to funnel plot asymmetry and heterogeneity. Under the recessive model, the summary estimate was POR = 0.93 (95% confidence interval: 0.70-1.23). We conclude that there is no or little association between DRD3 rs6280 polymorphisms and prevalence of TD.