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BAC-mediated gene-dosage analysis reveals a role for Zipro1 (Ru49/Zfp38) in progenitor cell proliferation in cerebellum and skin.

Genetic analysis in mice has most commonly employed two general strategies: phenotypic screens for spontaneous or induced mutations and genotypic analysis using homologous recombination or gene trapping to produce deletion or insertion mutants. Here we use bacterial artificial chromosome (BAC)-mediated gene-dosage analysis in transgenic mice to reveal novel genetic functions that are not evident from conventional loss-of-function mutations. We demonstrate a role for the zinc-finger transcription factor Zipro1 (formerly Ru49 and Zfp38) in the proliferation of granule cell precursors in the developing cerebellum, and document the contribution of this process to the final stages of cerebellar morphogenesis. We also show that Zipro1 is expressed in skin, and increased Zipro1 dosage results in a hair-loss phenotype associated with increased epithelial cell proliferation and abnormal hair follicle development.

Pubmed ID: 10431235

Authors

  • Yang XW
  • Wynder C
  • Doughty ML
  • Heintz N

Journal

Nature genetics

Publication Data

August 26, 1999

Associated Grants

  • Agency: NIGMS NIH HHS, Id: GM07739
  • Agency: DS NIH HHS, Id: NINDS 30532

Mesh Terms

  • Animals
  • Cell Count
  • Cell Death
  • Cell Division
  • Cerebellum
  • Chromosomes, Bacterial
  • DNA-Binding Proteins
  • Gene Dosage
  • Genetic Techniques
  • Hair
  • Mice
  • Mice, Transgenic
  • Models, Genetic
  • Phenotype
  • Skin
  • Stem Cells
  • Trans-Activators