2016
DOI: 10.1038/nn.4258
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Chd7 cooperates with Sox10 and regulates the onset of CNS myelination and remyelination

Abstract: Mutations in CHD7, encoding ATP-dependent chromodomain-helicase-DNA-binding protein 7, in CHARGE syndrome leads to multiple congenital anomalies including craniofacial malformations, neurological dysfunction and growth delay. Currently, mechanisms underlying the CNS phenotypes remain poorly understood. Here, we show that Chd7 is a direct transcriptional target of oligodendrogenesis-promoting factors Olig2 and Smarca4/Brg1, and is required for proper onset of CNS myelination and remyelination. Genome-occupancy … Show more

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Cited by 158 publications
(211 citation statements)
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“…CHD7 binds to thousands of promoter and regulatory regions in the mammalian genome (30) and has been shown to activate expression of several downstream target genes, including Twist1, Reelin, Sox4/Sox11, Otx2, Gbx2, Osterix, and Creb3l2 (39,(43)(44)(45)(46). To our knowledge, this is the first report of a confirmed target gene that is repressed by CHD7.…”
Section: Discussionmentioning
confidence: 84%
“…CHD7 binds to thousands of promoter and regulatory regions in the mammalian genome (30) and has been shown to activate expression of several downstream target genes, including Twist1, Reelin, Sox4/Sox11, Otx2, Gbx2, Osterix, and Creb3l2 (39,(43)(44)(45)(46). To our knowledge, this is the first report of a confirmed target gene that is repressed by CHD7.…”
Section: Discussionmentioning
confidence: 84%
“…The CHD family of proteins have pleotropic functions: chromatin remodeling, regulation of embryonic stem cell pluripotency, methylated histone binding, DNA binding, transcriptional regulation, cell cycle regulation, and regulation of apoptosis (He et al 2016; Manning and Yusufzai 2017; Zentner, Hurd, et al 2010). The vast majority of CHD7 mutations demonstrable in individuals with full CHARGE spectrum are truncating mutations (frameshift; non-sense) that are distributed across all 37 coding exons, suggesting haplo-insufficiency of CHD7 as the major molecular mechanism for this syndrome (Zentner, Layman, et al 2010).…”
Section: Molecular and Biologic Basis Of Endocrine Defects In Chargementioning
confidence: 99%
“…Furthermore, BRG1 chromatin remodeler is prepatterned with OLIG2 to facilitate expression of oligodendrocyte lineage-specific genes [27], functioning as a feed-forward loop, as the OLIG2/BRG1 complex further targets another chromatin remodeling enzyme, the ATP-dependent chromodomain helicase DNA-binding protein 7( Chd7 ). Genome-wide mapping of CHD7 target sites revealed that CHD7 forms complexes with SOX10 to activate positive regulatory oligodendrocyte genes, thereby initiating the myelination program [28]. Interestingly, BRG1-dependent chromatin remodeling is dispensable during the later stage of oligodendrocyte maturation, as Brg1 deletion following lineage specification did not affect OPC proliferation, migration, or survival, with only a mild defect in differentiation [29].…”
Section: Introductionmentioning
confidence: 99%