Frontal Bone Insufficiency in Gsk3β Mutant Mice

PloS One
Heather Szabo-RogersKaren J Liu

Abstract

The development of the mammalian skull is a complex process that requires multiple tissue interactions and a balance of growth and differentiation. Disrupting this balance can lead to changes in the shape and size of skull bones, which can have serious clinical implications. For example, insufficient ossification of the bony elements leads to enlarged anterior fontanelles and reduced mechanical protection of the brain. In this report, we find that loss of Gsk3β leads to a fully penetrant reduction of frontal bone size and subsequent enlarged frontal fontanelle. In the absence of Gsk3β the frontal bone primordium undergoes increased cell death and reduced proliferation with a concomitant increase in Fgfr2-IIIc and Twist1 expression. This leads to a smaller condensation and premature differentiation. This phenotype appears to be Wnt-independent and is not rescued by decreasing the genetic dose of β-catenin/Ctnnb1. Taken together, our work defines a novel role for Gsk3β in skull development.

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Citations

Mar 1, 2017·Journal of Anatomy·Brian J CusackHeather L Szabo-Rogers
Dec 24, 2018·Scientific Reports·Yong WanHeather L Szabo-Rogers
Jan 17, 2020·Genes·Brandi LantzHeather L Szabo-Rogers
Aug 22, 2018·Journal of Experimental Neuroscience·Sandra G Gonzalez Malagon, Karen J Liu
Apr 14, 2021·JCI Insight·Jiayi YaoYucheng Yao
Aug 24, 2021·Journal of Bone and Mineral Research : the Official Journal of the American Society for Bone and Mineral Research·Yong Wan, Heather L Szabo-Rogers

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Methods Mentioned

BETA
gene knockouts
X-ray
transgenic

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