First Author | Howng SY | Year | 2010 |
Journal | Genes Dev | Volume | 24 |
Issue | 3 | Pages | 301-11 |
PubMed ID | 20080941 | Mgi Jnum | J:156938 |
Mgi Id | MGI:4422117 | Doi | 10.1101/gad.1864510 |
Citation | Howng SY, et al. (2010) ZFP191 is required by oligodendrocytes for CNS myelination. Genes Dev 24(3):301-11 |
abstractText | The controlling factors that prompt mature oligodendrocytes to myelinate axons are largely undetermined. In this study, we used a forward genetics approach to identify a mutant mouse strain characterized by the absence of CNS myelin despite the presence of abundant numbers of late-stage, process-extending oligodendrocytes. Through linkage mapping and complementation testing, we identified the mutation as a single nucleotide insertion in the gene encoding zinc finger protein 191 (Zfp191), which is a widely expressed, nuclear-localized protein that belongs to a family whose members contain both DNA-binding zinc finger domains and protein-protein-interacting SCAN domains. Zfp191 mutants express an array of myelin-related genes at significantly reduced levels, and our in vitro and in vivo data indicate that mutant ZFP191 acts in a cell-autonomous fashion to disrupt oligodendrocyte function. Therefore, this study demonstrates that ZFP191 is required for the myelinating function of differentiated oligodendrocytes. |