Chordin Is a Modifier of Tbx1 for the Craniofacial Malformations of 22q11 Deletion Syndrome Phenotypes in Mouse
2009

Chordin as a Modifier of Tbx1 in Craniofacial Malformations

Sample size: 97 publication 10 minutes Evidence: moderate

Author Information

Author(s): Choi Murim, Klingensmith John, Morrow Bernice E.

Primary Institution: Duke University Medical Center

Hypothesis

Could an unidentified second-site lesion modify the craniofacial consequences of TBX1 deficiency?

Conclusion

Chordin is a modifier for the craniofacial anomalies of Tbx1 mutations, indicating a second-site modifier for specific phenotypes associated with 22q11 deletion syndrome.

Supporting Evidence

  • Chordin mutations in mice lead to defects similar to those seen in 22q11 deletion syndrome.
  • Chordin's effect on Tbx1 expression was observed in the pharyngeal region.
  • Different genetic backgrounds in mice resulted in varying penetrance of craniofacial defects.

Takeaway

This study found that a gene called chordin affects how another gene, Tbx1, causes facial problems in mice, showing that there are hidden factors that can change how these problems show up.

Methodology

The study involved breeding mice with specific genetic mutations and analyzing the resulting phenotypes.

Limitations

The study primarily focuses on mouse models, which may not fully replicate human conditions.

Participant Demographics

Mice of different genetic backgrounds were used in the study.

Statistical Information

P-Value

0.029

Statistical Significance

p=0.029

Digital Object Identifier (DOI)

10.1371/journal.pgen.1000395

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