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Magnetic resonance imaging (MRI) of BFPP brains reveals myelination defects in addition to brain malformation. However, the cellular role of GPR56 in oligodendrocyte development remains unknown. Here, we demonstrate that loss of\n                    <jats:italic>Gpr56<\/jats:italic>\n                    leads to hypomyelination of the central nervous system in mice. GPR56 levels are abundant throughout early stages of oligodendrocyte development, but are downregulated in myelinating oligodendrocytes.\n                    <jats:italic>Gpr56<\/jats:italic>\n                    -knockout mice manifest with decreased oligodendrocyte precursor cell (OPC) proliferation and diminished levels of active RhoA, leading to fewer mature oligodendrocytes and a reduced number of myelinated axons in the corpus callosum and optic nerves. Conditional ablation of\n                    <jats:italic>Gpr56<\/jats:italic>\n                    in OPCs leads to a reduced number of mature oligodendrocytes as seen in constitutive knockout of\n                    <jats:italic>Gpr56<\/jats:italic>\n                    . Together, our data define GPR56 as a cell-autonomous regulator of oligodendrocyte development.\n                  <\/jats:p>","DOI":"10.1038\/ncomms7121","type":"journal-article","created":{"date-parts":[[2015,1,21]],"date-time":"2015-01-21T09:22:10Z","timestamp":1421832130000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":139,"title":["The adhesion G protein-coupled receptor GPR56 is a cell-autonomous regulator of oligodendrocyte development"],"prefix":"10.1038","volume":"6","author":[{"given":"Stefanie","family":"Giera","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yiyu","family":"Deng","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rong","family":"Luo","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sarah D.","family":"Ackerman","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amit","family":"Mogha","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kelly R.","family":"Monk","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yanqin","family":"Ying","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sung-Jin","family":"Jeong","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Manabu","family":"Makinodan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Allison R.","family":"Bialas","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bernard S.","family":"Chang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Beth","family":"Stevens","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gabriel","family":"Corfas","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xianhua","family":"Piao","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2015,1,21]]},"reference":[{"key":"BFncomms7121_CR1","doi-asserted-by":"publisher","first-page":"871","DOI":"10.1152\/physrev.2001.81.2.871","volume":"81","author":"N Baumann","year":"2001","unstructured":"Baumann, N. & Pham-Dinh, D. 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