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

44 Citations (Scopus)

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
Externally publishedYes

Fingerprint

Oligodendroglia
Zinc Fingers
Myelin Sheath
Mutant Strains Mice
Insertional Mutagenesis
Chromosome Mapping
Mutant Proteins
Nuclear Proteins
Axons
Nucleotides
Mutation
DNA
Genes
Proteins

Keywords

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

ASJC Scopus subject areas

  • Genetics
  • Developmental Biology

Cite this

Howng, S. Y. B., Avila, R. L., Emery, B., Traka, M., Lin, W., Watkins, T., ... 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

ZFP191 is required by oligodendrocytes for CNS myelination. / Howng, Shen Yi B; Avila, Robin L.; Emery, Ben; Traka, Maria; Lin, Wensheng; Watkins, Trent; Cook, Susan; Bronson, Roderick; Davisson, Muriel; Barres, Ben A.; Popko, Brian.

In: Genes and Development, Vol. 24, No. 3, 01.02.2010, p. 301-311.

Research output: Contribution to journalArticle

Howng, SYB, Avila, RL, Emery, B, Traka, M, Lin, W, Watkins, T, Cook, S, Bronson, R, Davisson, M, Barres, BA & Popko, B 2010, 'ZFP191 is required by oligodendrocytes for CNS myelination', Genes and Development, vol. 24, no. 3, pp. 301-311. https://doi.org/10.1101/gad.1864510
Howng SYB, Avila RL, Emery B, Traka M, Lin W, Watkins T et al. ZFP191 is required by oligodendrocytes for CNS myelination. Genes and Development. 2010 Feb 1;24(3):301-311. https://doi.org/10.1101/gad.1864510
Howng, Shen Yi B ; Avila, Robin L. ; Emery, Ben ; Traka, Maria ; Lin, Wensheng ; Watkins, Trent ; Cook, Susan ; Bronson, Roderick ; Davisson, Muriel ; Barres, Ben A. ; Popko, Brian. / ZFP191 is required by oligodendrocytes for CNS myelination. In: Genes and Development. 2010 ; Vol. 24, No. 3. pp. 301-311.
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