ZFP191 is required by oligodendrocytes for CNS myelination.
Document Type
Article
Publication Date
2010
Keywords
Animals, Carrier-Proteins, Cell-Differentiation, Cell-Line, Cell-Proliferation, Cells-Cultured, Central-Nervous-System, Embryo-Mammalian, Mice-Transgenic, Mutation, Myelin-Sheath, Oligodendroglia
First Page
301
Last Page
311
JAX Source
Genes Dev 2010 Feb; 24(3):301-11.
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.
Recommended Citation
Howng SY,
Avila RL,
Emery B,
Traka M,
Lin W,
Watkins T,
Cook S,
Bronson R,
Davisson M,
Barres BA,
Popko B.
ZFP191 is required by oligodendrocytes for CNS myelination. Genes Dev 2010 Feb; 24(3):301-11.