First Author | Szabo-Rogers H | Year | 2016 |
Journal | PLoS One | Volume | 11 |
Issue | 2 | Pages | e0149604 |
PubMed ID | 26886780 | Mgi Jnum | J:336361 |
Mgi Id | MGI:6252895 | Doi | 10.1371/journal.pone.0149604 |
Citation | Szabo-Rogers H, et al. (2016) Frontal Bone Insufficiency in Gsk3beta Mutant Mice. PLoS One 11(2):e0149604 |
abstractText | 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 Gsk3beta leads to a fully penetrant reduction of frontal bone size and subsequent enlarged frontal fontanelle. In the absence of Gsk3beta 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 beta-catenin/Ctnnb1. Taken together, our work defines a novel role for Gsk3beta in skull development. |