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Recent developments have revealed a complex underlying panorama, including a spectrum of disorders in which parkinsonism is variably associated with additional neurological and non\u2010neurological signs. In particular, a childhood\u2010onset encephalopathy with epilepsy and\/or cognitive disability is the most common feature. Their genetic basis is also heterogeneous, with many causative genes and different mutation types ranging from \u201cclassical\u201d coding variants to intronic repeat expansions. In this review, we provide an updated overview of the phenotypic and genetic spectrum of the most relevant X\u2010linked parkinsonian syndromes, namely X\u2010linked dystonia\u2010parkinsonism (XDP, Lubag disease), fragile X\u2010associated tremor\/ataxia syndrome (FXTAS), beta\u2010propeller protein\u2010associated neurodegeneration (BPAN, NBIA\/PARK\u2010<jats:italic>WDR45<\/jats:italic>), Fabry disease, Waisman syndrome, methyl CpG\u2010binding protein 2 (MeCP2) spectrum disorder, phosphoglycerate kinase\u20101 deficiency syndrome (PGK1) and X\u2010linked parkinsonism and spasticity (XPDS<jats:italic>)<\/jats:italic>. All clinical and radiological features reported in the literature have been reviewed. Epilepsy occasionally represents the symptom of onset, predating parkinsonism even by a few years; action tremor is another common feature along with akinetic\u2010rigid parkinsonism. A focus on the genetic background and its pathophysiological implications is provided. The pathogenesis of these disorders ranges from well\u2010defined metabolic alterations (PGK1) to non\u2010specific lysosomal dysfunctions (XPDS) and vesicular trafficking alterations (Waisman syndrome). However, in other cases it still remains poorly defined. Recognition of the phenotypic and genetic heterogeneity of X\u2010linked parkinsonism has important implications for diagnosis, management, and genetic counseling. \u00a9 2021 The Authors. <jats:italic>Movement Disorders<\/jats:italic> published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society<\/jats:p>","DOI":"10.1002\/mds.28565","type":"journal-article","created":{"date-parts":[[2021,5,8]],"date-time":"2021-05-08T16:39:41Z","timestamp":1620491981000},"page":"1511-1525","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":24,"title":["X\u2010Linked Parkinsonism: Phenotypic and Genetic Heterogeneity"],"prefix":"10.1002","volume":"36","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2413-1788","authenticated-orcid":false,"given":"Giulia","family":"Di Lazzaro","sequence":"first","affiliation":[{"name":"Department of Clinical and Movement Neurosciences, UCL Queen Square Institute of 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