In a range of neurological conditions, including movement disorders, sex-related differences are emerging not only in brain anatomy and function, but also in pathogenesis, clinical features and response to treatment. In Parkinson disease (PD), for example, oestrogens can influence the severity of motor symptoms, whereas elevation of androgens can exacerbate tic disorders. Nevertheless, the real impact of sex differences in movement disorders remains under-recognized. In this article, we provide an up-to-date review of sex-related differences in PD and the most common hyperkinetic movement disorders, namely, essential tremor, dystonia, Huntington disease and other chorea syndromes, and Tourette syndrome and other chronic tic disorders. We highlight the most relevant clinical aspects of movement disorders that differ between men and women. Increased recognition of these differences and their impact on patient care could aid the development of tailored approaches to the management of movement disorders and enable the optimization of preclinical research and clinical studies.