Dystonia is a neurological disorder characterized by involuntary twisting movements and unnatural postures. There are many different forms of dystonia, which affect over three million people worldwide. Effective treatments are available only for a minority of patients, so new treatments are sorely needed. Several animal species have been used to develop models for different forms of dystonia, each with differing strengths and weaknesses. This review outlines the strategies that have been used to exploit these models for drug discovery. Some have been used to dissect the pathogenesis of dystonia for the identification of molecular targets for intervention. Others have been used for the empirical identification of candidate drugs. Therefore, the animal models provide promising new tools for developing better treatments for dystonia. © 2008 Informa UK Ltd.