Brain-derived neurotrophic factor (BDNF) is a modulator of synaptic and neural plasticity. Considering the association between dystonia and abnormal sensorimotor cortex plasticity, BDNF may be a candidate gene that confers susceptibility to dystonia. However, the association between Val66Met polymorphism of BDNF gene and primary dystonia is controversial.A case–control study was performed to evaluate the association between Val66Met polymorphism in the BDNF gene and primary dystonia in a cohort of 252 Chinese patients and in 214 age- and gender-matched healthy control subjects.No association was identified between Val66Met polymorphism and primary dystonia or cervical dystonia (P=0.309 and P=0.803 respectively). In a subsequent subgroup analysis, there was also no difference in the distribution for age of onset.Our findings do not support that BDNF Val66Met polymorphism contributes to the risk of primary dystonia.