Frontal Bone Insufficiency in Gsk3β Mutant Mice

Szabo-Rogers, Heather L.; Yakob, Wardati; Liu, Karen

doi:10.1371/journal.pone.0149604

Cited by 13 publications

(17 citation statements)

References 60 publications

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“…Molecularly, we observe decreased Wnt/β-catenin in the Bj mutant frontal bone primordium at E12.5. In agreement with previous reports regarding differences between the frontal and parietal bones 27,40 , we observe a defect only in the Prickle1 Bj/Bj frontal bone. Our data provides more support to the hypothesis that the parietal bone is not sensitive to changes in Wnt/β-catenin signaling and therefore develops normally in Prickle1 Bj/Bj .…”

Section: Discussionsupporting

confidence: 93%

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Prickle1 regulates differentiation of frontal bone osteoblasts

Wan

Lantz

Cusack

et al. 2018

Sci Rep

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Enlarged fontanelles and smaller frontal bones result in a mechanically compromised skull. Both phenotypes could develop from defective migration and differentiation of osteoblasts in the skull bone primordia. The Wnt/Planar cell polarity (Wnt/PCP) signaling pathway regulates cell migration and movement in other tissues and led us to test the role of Prickle1, a core component of the Wnt/PCP pathway, in the skull. For these studies, we used the missense allele of Prickle1 named Prickle1Beetlejuice (Prickle1Bj). The Prickle1Bj/Bj mutants are microcephalic and develop enlarged fontanelles between insufficient frontal bones, while the parietal bones are normal. Prickle1Bj/Bj mutants have several other craniofacial defects including a midline cleft lip, incompletely penetrant cleft palate, and decreased proximal-distal growth of the head. We observed decreased Wnt/β-catenin and Hedgehog signaling in the frontal bone condensations of the Prickle1Bj/Bj mutants. Surprisingly, the smaller frontal bones do not result from defects in cell proliferation or death, but rather significantly delayed differentiation and decreased expression of migratory markers in the frontal bone osteoblast precursors. Our data suggests that Prickle1 protein function contributes to both the migration and differentiation of osteoblast precursors in the frontal bone.

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Section: Discussionsupporting

confidence: 93%

“…Frontal bone insufficiency can result from defects in proliferation and cell death 27 . We undertook these studies at E12.5 because this is a critical stage for setting up the frontal bone condensation.…”

Section: Resultsmentioning

confidence: 99%

Prickle1 regulates differentiation of frontal bone osteoblasts

Wan

Lantz

Cusack

et al. 2018

Sci Rep

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“…; Szabo‐Rogers et al. ). BIO (6‐bromoindirubin 3′oxime) binds to the ATP binding pocket of both GSK‐3α and GSK‐3β, and prevents its activity (Meijer et al.…”

Section: Resultsmentioning

confidence: 99%

“…Inhibition of GSK-3 results in a discontinuous ethmoid plate GSK-3 is a promiscuous kinase that functions in multiple signaling pathways, including the Wnt, HH and NFAT/calcineurin pathways; however, GSK-3 function is most well known for its role in the b-catenin destruction complex (Doble & Woodgett, 2003). GSK-3 is a required anteriorizing factor in Xenopus, and is required for palatogenesis and skull development in the mouse (Liu et al 2007;Szabo-Rogers et al 2016). BIO (6-bromoindirubin 3 0 oxime) binds to the ATP binding pocket of both GSK-3a and GSK-3b, and prevents its activity (Meijer et al 2003).…”

Section: Gross Morphological Changes Resulting From Growth Factor Inhmentioning

confidence: 99%

Growth factor signaling alters the morphology of the zebrafish ethmoid plate

et al. 2017

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Craniofacial development relies on coordinated tissue interactions that allow for patterning and growth of the face. We know a priori that the Wingless, fibroblast growth factor, Hedgehog and transforming growth factor-beta growth factor signaling pathways are required for the development of the face, but how they contribute to the shape of the face is largely untested. Here, we test how each signaling pathway contributes to the overall morphology of the zebrafish anterior neurocranium. We tested the contribution of each signaling pathway to the development of the ethmoid plate during three distinct time periods: the time of neural crest migration [10 hour post fertilization (hpf)]; once the neural crest is resident in the face (20 hpf); and finally at the time at which the cartilaginous condensations are being initiated (48 hpf). Using geometric morphometric analysis, we conclude that each signaling pathway contributes to the shape, size and morphology of the ethmoid plate in a dose-, and time-dependent fashion.

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“…It was previously believed that neural crest cells migrate on top of mesoderm cells or that their migration is caused by the dynamics of surrounding ectoderm and mesoderm cells [ 24 ]. However, recent in vivo time-lapse imaging in chick embryos revealed that neural crest cells squeeze between ectoderm and mesoderm cells, but do not go over the top of these other cells [ 25 ].…”

Section: Neural Crest Cellsmentioning

confidence: 99%

Cranial Neural Crest Cells and Their Role in the Pathogenesis of Craniofacial Anomalies and Coronal Craniosynostosis

Siismets

Hatch

2020

JDB

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Craniofacial anomalies are among the most common of birth defects. The pathogenesis of craniofacial anomalies frequently involves defects in the migration, proliferation, and fate of neural crest cells destined for the craniofacial skeleton. Genetic mutations causing deficient cranial neural crest migration and proliferation can result in Treacher Collins syndrome, Pierre Robin sequence, and cleft palate. Defects in post-migratory neural crest cells can result in pre- or post-ossification defects in the developing craniofacial skeleton and craniosynostosis (premature fusion of cranial bones/cranial sutures). The coronal suture is the most frequently fused suture in craniosynostosis syndromes. It exists as a biological boundary between the neural crest-derived frontal bone and paraxial mesoderm-derived parietal bone. The objective of this review is to frame our current understanding of neural crest cells in craniofacial development, craniofacial anomalies, and the pathogenesis of coronal craniosynostosis. We will also discuss novel approaches for advancing our knowledge and developing prevention and/or treatment strategies for craniofacial tissue regeneration and craniosynostosis.

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Frontal Bone Insufficiency in Gsk3β Mutant Mice

Cited by 13 publications

References 60 publications

Prickle1 regulates differentiation of frontal bone osteoblasts

Prickle1 regulates differentiation of frontal bone osteoblasts

Growth factor signaling alters the morphology of the zebrafish ethmoid plate

Cranial Neural Crest Cells and Their Role in the Pathogenesis of Craniofacial Anomalies and Coronal Craniosynostosis

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