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Related Experiment Video

Updated: Apr 18, 2026

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Sufu and Kif7 in limb patterning and development.

Olena Zhulyn1, Chi-Chung Hui

  • 1Program in Developmental and Stem Cell Biology, The Hospital for Sick Children, Toronto, Ontario, Canada; Department of Molecular Genetics, University of Toronto, Toronto, Ontario, Canada.

Developmental Dynamics : an Official Publication of the American Association of Anatomists
|January 13, 2015
PubMed
Summary
This summary is machine-generated.

Loss of Sufu and Kif7 disrupts Sonic hedgehog (Shh) signaling, impacting Gli activity and causing severe limb defects. This study clarifies the roles of these key regulators in vertebrate digit patterning.

Keywords:
Gli transcription factorsKif7Sonic hedgehogSufulimb patterningtwo-population model

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Area of Science:

  • Developmental biology
  • Molecular genetics

Background:

  • Vertebrate digit pattern is regulated by Sonic hedgehog (Shh) morphogen.
  • Shh controls Gli transcription factors, with Sufu and Kif7 modulating their activity.
  • The specific roles of Sufu and Kif7 in limb development require further elucidation.

Purpose of the Study:

  • To investigate the roles of Sufu and Kif7 in limb development.
  • To analyze the effects of Sufu and Kif7 loss on Gli activity and digit patterning.

Main Methods:

  • Utilized a series of mouse mutants lacking Sufu and Kif7.
  • Observed Shh signaling pathway activity and limb formation in these mutants.

Main Results:

  • Sufu and Kif7 are expressed throughout limb development.
  • Deletion of Sufu and Kif7 leads to distinct effects on Gli levels and limb formation.
  • Concomitant deletion caused constitutive Shh pathway activity and severe limb truncation, consistent with the two-population model.

Conclusions:

  • Perturbations of Sufu and Kif7 significantly affect Gli activity.
  • Loss of Sufu and Kif7 recapitulates the spectrum of vertebrate limb defects, from truncation to polydactyly.