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Developmental mechanisms underlying polydactyly in the mouse mutant Doublefoot.

Alexandra P Crick1, Christian Babbs, Jennifer M Brown

  • 1Department of Human Anatomy and Genetics, South Parks Road, Oxford OX1 3QX, UK.

Journal of Anatomy
|February 18, 2003
PubMed
Summary

The Doublefoot mouse mutant exhibits polydactyly and altered limb polarity. New findings suggest its mutation is epistatic to Sonic hedgehog (Shh) signaling, impacting limb development.

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Area of Science:

  • Developmental biology
  • Genetics
  • Mouse models

Background:

  • The Doublefoot (Dbf) mouse mutant displays pre-axial polydactyly, characterized by 6-9 digits per limb and a lack of anteroposterior (AP) polarity.
  • Unlike other polydactylous mutants, Dbf shows normal Sonic hedgehog (Shh) expression, yet polarizing and hedgehog signaling activities are detected throughout the distal mesenchyme.

Purpose of the Study:

  • To review current understanding of the Doublefoot mouse mutant.
  • To present new findings on the genetic basis and developmental mechanisms underlying Dbf-associated limb abnormalities.

Main Methods:

  • Review of existing literature on the Doublefoot mutant.
  • Mouse-chick grafting experiments to assess polarizing activity.
  • Analysis of gene expression patterns, including Patched 1 (Ptc1) for hedgehog signaling.

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  • Investigation of limb bud expansion, cell proliferation rates, and ectopic gene expression.

    • Main Results:

    • Limb bud expansion in Dbf mutants is associated with ectopic Shh expression.
    • High cell proliferation rates, typically posterior, extend into the anterior limb bud region.
    • The Dbf mutation functions epistatically to Shh in limb development, indicating a regulatory relationship upstream of or parallel to Shh signaling.

    Conclusions:

    • The Doublefoot mutant provides a unique model for studying limb development and polarity.
    • Ectopic Shh expression and altered cell proliferation contribute to the polydactyly and polarity defects observed in Dbf mutants.
    • The Dbf mutation's epistasis to Shh suggests a critical role in regulating downstream signaling pathways essential for normal limb patterning.