Fuz Regulates Craniofacial Development through Tissue Specific Responses to Signaling Factors
Author Information
Author(s): Zhang Zichao, Wlodarczyk Bogdan J., Niederreither Karen, Venugopalan Shankar, Florez Sergio, Finnell Richard H., Amendt Brad A.
Primary Institution: Institute of Biosciences and Technology, Texas A&M Health Science Center
Hypothesis
Fuz plays a critical role in craniofacial development by regulating Hedgehog and Wnt signaling pathways.
Conclusion
Fuz is essential for proper craniofacial development, and its absence leads to severe deformities due to disrupted signaling pathways.
Supporting Evidence
- Fuz−/− mice exhibited severe craniofacial deformities including anophthalmia and cleft palate.
- Hedgehog signaling was down-regulated in Fuz null mice, while Wnt signaling was up-regulated.
- Fuz expression was shown to regulate both Hedgehog and Wnt signaling pathways during development.
- Chromatin IP experiments demonstrated that β-catenin directly regulates Fuz expression.
Takeaway
Fuz is like a traffic cop for signals that help shape the face; without it, the face can grow all wrong.
Methodology
The study used Fuz−/− mice to analyze craniofacial phenotypes and signaling responses, employing techniques like immunofluorescence and real-time PCR.
Limitations
The study primarily focused on mouse models, which may not fully represent human craniofacial development.
Statistical Information
P-Value
p<0.01
Statistical Significance
p<0.05
Digital Object Identifier (DOI)
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