Krause C, Schaake S, Grütz K, et al. - The role of DNA methylation in the pathogenesis of X‐linked dystonia‐parkinsonism was evaluated in this study. In DNA from peripheral blood leukocytes, fibroblasts, induced pluripotent stem cell–derived cortical neurons and brain tissue from X‐linked dystonia‐parkinsonism patients and age‐ and sex‐matched healthy Filipino controls, DNA methylation at DSC12, DSC3, and DSC2 was quantified by bisulfite pyrosequencing in this prospective study. Altered DNA methylation in patients with X‐linked dystonia‐parkinsonism was deemed as a possible additional mechanism modulating TAF1 expression and putative novel targets for future therapies utilizing DNA methylation‐modifying agents.