In a recent issue of Experimental Neurology, Prudente et al. (2012) investigated the neuropathology of cervical dystonia in six patients. Their most important finding was a patchy loss of cerebellar Purkinje cells in the cerebellum. In this article we discuss their findings in the context of a review including primary and secondary cervical dystonia. An update is given of the current knowledge on structural and functional brain abnormalities in idiopathic cervical dystonia with a special focus on the cerebellum.