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Mutations in genes involved in the glycosylation of dystroglycan cause a form of congenital muscular dystrophy termed dystroglycanopathy. In addition to its well\u2010defined role in regulating muscle integrity, dystroglycan is essential for proper central and peripheral nervous system development. Patients with dystroglycanopathy can present with a wide range of neurological perturbations, but unraveling the complex role of Dag1 in the nervous system has proven to be a challenge. Over the past two decades, animal models of dystroglycanopathy have been an invaluable resource that has allowed researchers to elucidate dystroglycan's many roles in neural circuit development. In this review, we summarize the pathways involved in dystroglycan's glycosylation and its known interacting proteins, and discuss how it regulates neuronal migration, axon guidance, synapse formation, and its role in non\u2010neuronal cells.<\/jats:p>","DOI":"10.1002\/dvdy.516","type":"journal-article","created":{"date-parts":[[2022,6,30]],"date-time":"2022-06-30T08:15:16Z","timestamp":1656576916000},"page":"61-80","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":47,"title":["The many roles of dystroglycan in nervous system development and function"],"prefix":"10.1002","volume":"252","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2319-6109","authenticated-orcid":false,"given":"Jennifer N.","family":"Jahncke","sequence":"first","affiliation":[{"name":"Neuroscience Graduate Program, Oregan Health &amp; Science University  Portland Oregon 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