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Updated: Feb 22, 2026

Analyzing Craniofacial Morphogenesis in Zebrafish Using 4D Confocal Microscopy
Published on: January 30, 2014
Dysregulated PDGFRα signaling alters coronal suture morphogenesis and leads to craniosynostosis through endochondral
Fenglei He1,2, Philippe Soriano3
1Department of Cell, Developmental and Regenerative Biology, Icahn School of Medicine at Mount Sinai, New York, NY 10029, USA.
Abstract:
Craniosynostosis is a prevalent human birth defect characterized by premature fusion of calvarial bones. In this study, we show that tight regulation of endogenous PDGFRα activity is required for normal calvarium development in the mouse and that dysregulated PDGFRα activity causes craniosynostosis. Constitutive activation of PDGFRα leads to expansion of cartilage underlying the coronal sutures, which contribute to suture closure through endochondral ossification, in a process regulated in part by PI3K/AKT signaling. Our results thus identify a novel mechanism underlying calvarial development in craniosynostosis.
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