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During early embryonic development, motor neurons and then oligodendrocyte precursor cells (OPCs) are specified from neural progenitors residing in the periventricular pMN progenitor domain of the spinal cord. Following gliogenesis, OPCs can differentiate as oligodendrocytes (OLs)\u2014the myelinating glial cells of the central nervous system\u2014or remain as OPCs. To generate unique cell types capable of highly divergent functions, these specification and differentiation events require specialized gene expression programs. RNA binding proteins (RBPs) regulate mRNA localization and translation in the developing nervous system and are linked to many neurodevelopmental disorders. One example is Fragile X syndrome (FXS), caused by the loss of the RBP fragile X mental retardation protein (FMRP). Importantly, infants with FXS have reduced white matter and we previously showed that zebrafish Fmrp is autonomously required in OLs to promote myelin sheath growth. We now find that Fmrp regulates cell specification in pMN progenitor cells such that\n                    <jats:italic>fmr1<\/jats:italic>\n                    mutant zebrafish generate fewer motor neurons and excess OPCs. Fmrp subsequently promotes differentiation of OPCs, leading to fewer differentiating OLs in the developing spinal cord of\n                    <jats:italic>fmr1<\/jats:italic>\n                    larvae. Although the early patterning of spinal progenitor domains appears largely normal in\n                    <jats:italic>fmr1<\/jats:italic>\n                    mutants during early embryogenesis, Shh signaling is greatly diminished. Taken together, these results suggest cell stage\u2010specific requirements for Fmrp in the specification and differentiation of oligodendrocyte lineage cells.\n                  <\/jats:p>","DOI":"10.1002\/glia.24041","type":"journal-article","created":{"date-parts":[[2021,6,10]],"date-time":"2021-06-10T18:45:54Z","timestamp":1623350754000},"page":"2349-2361","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":15,"title":["Fmrp regulates oligodendrocyte lineage cell specification and differentiation"],"prefix":"10.1002","volume":"69","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2109-2448","authenticated-orcid":false,"given":"Caleb A.","family":"Doll","sequence":"first","affiliation":[{"name":"Department of Pediatrics, Section of Developmental Biology University of Colorado School of Medicine, Children's Hospital Colorado  Aurora Colorado USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kayt","family":"Scott","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Section of Developmental Biology University of Colorado School of Medicine, Children's Hospital Colorado  Aurora Colorado USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bruce","family":"Appel","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Section of Developmental Biology University of Colorado School of Medicine, Children's Hospital Colorado  Aurora Colorado USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2021,6,10]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.101717"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature11737"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1038\/nmeth.4509"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.38205"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1038\/35012083"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.124.22.4557"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0092-8674(00)80853-3"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.122.9.2835"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2015.06.017"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2011.06.013"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1044-7431(03)00210-0"},{"key":"e_1_2_9_13_1","first-page":"2","article-title":"Generation and characterization of Fmr1 knockout zebrafish","volume":"4","author":"Broeder M. 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