ZFP191 is required by oligodendrocytes for CNS myelination

Shen Yi B. Howng, Robin L. Avila, Ben Emery, Maria Traka, Wensheng Lin, Trent Watkins, Susan Cook, Roderick Bronson, Muriel Davisson, Ben A. Barres, Brian Popko

Research output: Contribution to journalArticle

49 Scopus citations

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-proteininteracting 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.

Original languageEnglish (US)
Pages (from-to)301-311
Number of pages11
JournalGenes and Development
Volume24
Issue number3
DOIs
StatePublished - Feb 1 2010

Keywords

  • Conditional allele
  • Forward genetics
  • Hypomyelination
  • Mouse mutant
  • Scan domain
  • Zinc finger protein

ASJC Scopus subject areas

  • Genetics
  • Developmental Biology

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    Howng, S. Y. B., Avila, R. L., Emery, B., Traka, M., Lin, W., Watkins, T., Cook, S., Bronson, R., Davisson, M., Barres, B. A., & Popko, B. (2010). ZFP191 is required by oligodendrocytes for CNS myelination. Genes and Development, 24(3), 301-311. https://doi.org/10.1101/gad.1864510