ArticleHuman molecular genetics2013
Quantification of shape and cell polarity reveals a novel mechanism underlying malformations resulting from related FGF mutations during facial morphogenesis.
Article in Human molecular genetics, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 22 papers.
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Who cites it
22 citing papers in PubMed, 36 citations in OpenAlex.
- Quantification of facial skeletal shape variation in fibroblast growth factor receptor-related craniosynostosis syndromes.Birth defects research. Part A, Clinical and molecular teratology · 2014Trial
- Navigating the window: structural bottlenecks in avian craniofacial integration revealed by the ontogenetic patterning of shape variance.bioRxiv : the preprint server for biology · 2026Article
- Three-dimensional cellular dynamics and mandibular morphogenesis.Frontiers in cell and developmental biology · 2026Article
- Cell jamming transitions can affect regulatory protein gradients and prime evolutionary divergence.Journal of the Royal Society, Interface · 2025Article
- Downstream branches of receptor tyrosine kinase signaling act interdependently to shape the face.Developmental dynamics : an official publication of the American Association of Anatomists · 2025Article
- Dosage-dependent effects of FGFR2Anatomical record (Hoboken, N.J. : 2007) · 2025Article
- Quantifying the relationship between cell proliferation and morphology during development of the face.Development (Cambridge, England) · 2025Article
- Zebrafish crocc2 mutants exhibit divergent craniofacial shape, misregulated variability, and aberrant cartilage morphogenesis.Developmental dynamics : an official publication of the American Association of Anatomists · 2023Article
- Phenotypic variability of syndromic craniosynostosis caused by c.833G > T in FGFR2: Clinical and genetic evaluation of eight patients from a five-generation family.Molecular genetics & genomic medicine · 2022Article
- Review
- Aberrant cell segregation in the craniofacial primordium and the emergence of facial dysmorphology in craniofrontonasal syndrome.PLoS genetics · 2020Article
- Association of IFT88 gene variants with nonsyndromic cleft lip with or without cleft palate.Birth defects research · 2019Article
- Molecular and cellular mechanisms underlying the evolution of form and function in the amniote jaw.EvoDevo · 2019Review
- FGF controls epithelial-mesenchymal transitions during gastrulation by regulating cell division and apicobasal polarity.Development (Cambridge, England) · 2018Article
- Neural crest cells utilize primary cilia to regulate ventral forebrain morphogenesis via Hedgehog-dependent regulation of oriented cell division.Developmental biology · 2017Article
- Beyond cell proliferation in avian facial morphogenesis.Developmental dynamics : an official publication of the American Association of Anatomists · 2016Review
- Molecular mechanisms of midfacial developmental defects.Developmental dynamics : an official publication of the American Association of Anatomists · 2016Review
- Fibroblast growth factor signaling in myofibroblasts differs from lipofibroblasts during alveolar septation in mice.American journal of physiology. Lung cellular and molecular physiology · 2015Article
- A dynamic Shh expression pattern, regulated by SHH and BMP signaling, coordinates fusion of primordia in the amniote face.Development (Cambridge, England) · 2015Article
- Tfap2a-dependent changes in mouse facial morphology result in clefting that can be ameliorated by a reduction in Fgf8 gene dosage.Disease models & mechanisms · 2015Article
Corrections and comments
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Authors and funding
7 authors at 3 institutions in 2 countries.
Funding
Abstract
Fibroblast growth factor (FGF) signaling mutations are a frequent contributor to craniofacial malformations including midfacial anomalies and craniosynostosis. FGF signaling has been shown to control cellular mechanisms that contribute to facial morphogenesis and growth such as proliferation, survival, migration and differentiation. We hypothesized that FGF signaling not only controls the magnitude of growth during facial morphogenesis but also regulates the direction of growth via cell polarity. To test this idea, we infected migrating neural crest cells of chicken embryos with replication-competent avian sarcoma virus expressing either FgfR2(C278F), a receptor mutation found in Crouzon syndrome or the ligand Fgf8. Treated embryos exhibited craniofacial malformations resembling facial dysmorphologies in craniosynostosis syndrome. Consistent with our hypothesis, ectopic activation of FGF signaling resulted in decreased cell proliferation, increased expression of the Sprouty class of FGF signaling inhibitors, and repressed phosphorylation of ERK/MAPK. Furthermore, quantification of cell polarity in facial mesenchymal cells showed that while orientation of the Golgi body matches the direction of facial prominence outgrowth in normal cells, in FGF-treated embryos this direction is randomized, consistent with aberrant growth that we observed. Together, these data demonstrate that FGF signaling regulates cell proliferation and cell polarity and that these cell processes contribute to facial morphogenesis.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.