Format

Send to

Choose Destination
Genes Dev. 2010 Feb 1;24(3):301-11. doi: 10.1101/gad.1864510. Epub 2010 Jan 15.

ZFP191 is required by oligodendrocytes for CNS myelination.

Author information

1
Department of Human Genetics, The University of Chicago, Chicago, Illinois 60637, USA;

Abstract

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.

PMID:
20080941
PMCID:
PMC2811831
DOI:
10.1101/gad.1864510
[Indexed for MEDLINE]
Free PMC Article

Supplemental Content

Full text links

Icon for HighWire Icon for PubMed Central
Loading ...
Support Center