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Although Ermin has been predicted to play a role in the formation and stability of myelin sheaths, this has not been directly examined in vivo. Here, we show that Ermin is essential for myelin sheath integrity and normal saltatory conduction. Loss of Ermin in mice caused de\u2010compacted and fragmented myelin sheaths and led to slower conduction along with progressive neurological deficits. RNA sequencing of the corpus callosum, the largest white matter structure in the CNS, pointed to inflammatory activation in aged Ermin\u2010deficient mice, which was corroborated by increased levels of microgliosis and astrogliosis. The inflammatory milieu and myelin abnormalities were further associated with increased susceptibility to immune\u2010mediated demyelination insult in Ermin knockout mice. Supporting a possible role of Ermin deficiency in inflammatory white matter disorders, a rare inactivating mutation in the\n                    <jats:italic>ERMN<\/jats:italic>\n                    gene was identified in multiple sclerosis patients. Our findings demonstrate a critical role for Ermin in maintaining myelin integrity. Given its near\u2010exclusive expression in myelinating oligodendrocytes, Ermin deficiency represents a compelling \u201cinside\u2010out\u201d model of inflammatory dysmyelination and may offer a new paradigm for the development of myelin stability\u2010targeted therapies.\n                  <\/jats:p>","DOI":"10.1111\/bpa.13064","type":"journal-article","created":{"date-parts":[[2022,3,14]],"date-time":"2022-03-14T02:13:58Z","timestamp":1647224038000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":18,"title":["Ermin deficiency leads to compromised myelin, inflammatory milieu, and susceptibility to demyelinating insult"],"prefix":"10.1111","volume":"32","author":[{"given":"Amin","family":"Ziaei","sequence":"first","affiliation":[{"name":"Translational Laboratory in Genetic Medicine (TLGM) Agency for Science, Technology, and Research (A*STAR)  Singapore Singapore"},{"name":"UCSF Weill Institute for Neurosciences University of 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