Defining a critical period in calvarial development for Hedgehog pathway antagonist-induced frontal bone dysplasia in mice

Abstract

During embryonic development, the Hedgehog signalling pathway regulates the migration, proliferation and differentiation of cranial neural crest cells in the early frontal bone. The Hedgehog signalling pathway transmits information to embryonic cells for their proper cell differentiation, and increased or reduced function of that signalling results in various craniofacial malformations. A team headed by Weihui Chen at Fujian Medical University in China investigated the patterns of abnormalities caused by inhibition of Hedgehog signalling in pregnant mice at preselected embryonic time points. The team was able to identify the critical period for sensitivity to GDC-0449, a potent Hedgehog signalling inhibitor. The authors believe that their mouse model can be effective in further investigating the mechanisms of craniofacial malformations and will have a profound impact on identifying candidate human disease genes and associated environmental factors.

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Funding

National Natural Science Foundation of China (National Science Foundation of China)(31070838, 31070838, 31070838, 31070838, 31070838)