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This perspective presents the developing stories of several childhood\u2010onset movement disorders, including paroxysmal kinesigenic dyskinesia, myoclonus\u2010dystonia syndrome, and other monogenic dystonias. These stories illustrate how gene discovery helps focus the research efforts of scientists trying to understand the mechanisms of disease. The genetic diagnosis of these clinical syndromes also helps clarify the associated phenotypic spectra and aids the search for additional disease\u2010causing genes. Collectively, the findings of previous studies have led to increased recognition of the role of the cerebellum in the physiology and pathophysiology of motor control\u2014a common theme in many pediatric movement disorders. To fully exploit the genetic information garnered in the clinical and research arenas, it is crucial that corresponding multi\u2010omics analyses and functional studies also be performed at scale. Hopefully, these integrated efforts will provide us with a more comprehensive understanding of the genetic and neurobiological bases of movement disorders in childhood.<\/jats:p>","DOI":"10.1002\/ggn2.202200018","type":"journal-article","created":{"date-parts":[[2022,11,9]],"date-time":"2022-11-09T20:34:31Z","timestamp":1668026071000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Translating Genetic Discovery into a Mechanistic Understanding of Pediatric Movement Disorders: Lessons from Genetic Dystonias and Related Disorders"],"prefix":"10.1002","volume":"4","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-9016-2067","authenticated-orcid":false,"given":"Wei\u2010Sheng","family":"Lin","sequence":"first","affiliation":[{"name":"Department of Pediatrics Taipei Veterans General Hospital  Taipei 11217 Taiwan"},{"name":"School of Medicine National Yang Ming Chiao Tung University  Taipei 112304 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