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Neural tube, skeletal and body wall defects in mice lacking transcription factor AP-2

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Abstract

THE retinoic acid-inducible transcription factor AP-2 is expressed in epithelial and neural crest cell lineages during murine development1–5. AP-2 can regulate neural and epithelial gene transcription, and is associated with overexpression of c-erbB-2 in human breast-cancer cell lines4–6. To ascertain the importance of AP-2 for normal development, we have derived mice containing a homozygous disruption of the AP-2 gene. These AP-2-null mice have multiple congenital defects and die at birth. In particular, the AP-2 knockout mice exhibit anencephaly, craniofacial defects and thoraco-abdominoschisis. Skeletal defects occur in the head and trunk region, where many bones are deformed or absent. Analysis of these mice earlier in embryogenesis indicates a failure of cranial neural-tube closure and defects in cranial ganglia development. We have shown that AP-2 is a fundamental regulator of mammalian craniofacial development.

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Authors and Affiliations

  1. Department of Biology, Yale University, 266 Whitney Avenue, New Haven, Connecticut, 06511, USA

    Jian Zhang, Stephanie Hagopian-Donaldson, Debora Plehn-Dujowich & Trevor Williams

  2. Department of Cellular and Developmental Biology, The Biological Laboratories, Harvard University, 16 Divinity Avenue, Cambridge, Massachusetts, 02138, USA

    George Serbedzija & Andrew P. McMahon

  3. Howard Hughes Medical Institute and Section of Immunobiology, Yale University School of Medicine, New Haven, Connecticut, 06510, USA

    Jennifer Elsemore & Richard A. Flavell

Authors
  1. Jian Zhang
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  2. Stephanie Hagopian-Donaldson
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  3. George Serbedzija
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  4. Jennifer Elsemore
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  5. Debora Plehn-Dujowich
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  6. Andrew P. McMahon
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  7. Richard A. Flavell
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  8. Trevor Williams
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Zhang, J., Hagopian-Donaldson, S., Serbedzija, G. et al. Neural tube, skeletal and body wall defects in mice lacking transcription factor AP-2. Nature 381, 238–241 (1996). https://doi.org/10.1038/381238a0

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  • DOI: https://doi.org/10.1038/381238a0

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