ArticleDevelopmental biology2014
Directed Bmp4 expression in neural crest cells generates a genetic model for the rare human bony syngnathia birth defect.
Article in Developmental biology, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers.
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Who cites it
18 citing papers in PubMed, 45 citations in OpenAlex.
- Effects of Smoothened Agonist Exposure on Murine Craniofacial Development.International dental journal · 2026Article
- Article
- Transcriptome sequencing of facial adipose tissue reveals alterations in mRNAs of hemifacial microsomia.Frontiers in pediatrics · 2023Article
- TWIST1 interacts with β/δ-catenins during neural tube development and regulates fate transition in cranial neural crest cells.Development (Cambridge, England) · 2022Article
- Article
- The effects of altered BMP4 signaling in first branchial-arch-derived murine embryonic orofacial tissues.International journal of oral science · 2021Article
- GATA3 is essential for separating patterning domains during facial morphogenesis.Development (Cambridge, England) · 2021Article
- Augmented BMP4 signal impairs tongue myogenesis.Journal of molecular histology · 2021Article
- The Mandibular and Hyoid Arches-From Molecular Patterning to Shaping Bone and Cartilage.International journal of molecular sciences · 2021Review
- Congenital Maxillomandibular Syngnathia: Review of Literature and Proposed New Classification System.Journal of maxillofacial and oral surgery · 2021Article
- AP-2α and AP-2β cooperatively orchestrate homeobox gene expression during branchial arch patterning.Development (Cambridge, England) · 2018Article
- Intercellular Genetic Interaction Between Irf6 and Twist1 during Craniofacial Development.Scientific reports · 2017Article
- Exclusion of Dlx5/6 expression from the distal-most mandibular arches enables BMP-mediated specification of the distal cap.Proceedings of the National Academy of Sciences of the United States of America · 2016Article
- Common mechanisms in development and disease: BMP signaling in craniofacial development.Cytokine & growth factor reviews · 2016Review
- The short stature homeobox 2 (Shox2)-bone morphogenetic protein (BMP) pathway regulates dorsal mesenchymal protrusion development and its temporary function as a pacemaker during cardiogenesis.The Journal of biological chemistry · 2015Article
- Mouse Models of Rare Craniofacial Disorders.Current topics in developmental biology · 2015Review
- BMP-FGF signaling axis mediates Wnt-induced epidermal stratification in developing mammalian skin.PLoS genetics · 2014Article
- Application values of prenatal screening and non-invasive gene sequencing in fetal birth defects.Pakistan journal of medical sciencesArticle
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Authors and funding
10 authors at 4 institutions in 2 countries.
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Abstract
Congenital bony syngnathia, a rare but severe human birth defect, is characterized by bony fusion of the mandible to the maxilla. However, the genetic mechanisms underlying this birth defect are poorly understood, largely due to limitation of available animal models. Here we present evidence that transgenic expression of Bmp4 in neural crest cells causes a series of craniofacial malformations in mice, including a bony fusion between the maxilla and hypoplastic mandible, resembling the bony syngnathia syndrome in humans. In addition, the anterior portion of the palatal shelves emerged from the mandibular arch instead of the maxilla in the mutants. Gene expression assays showed an altered expression of several facial patterning genes, including Hand2, Dlx2, Msx1, Barx1, Foxc2 and Fgf8, in the maxillary and mandibular processes of the mutants, indicating mis-patterned cranial neural crest (CNC) derived cells in the facial region. However, despite of formation of cleft palate and ectopic cartilage, forced expression of a constitutively active form of BMP receptor-Ia (caBmprIa) in CNC lineage did not produce the syngnathia phenotype, suggesting a non-cell autonomous effect of the augmented BMP4 signaling. Our studies demonstrate that aberrant BMP4-mediated signaling in CNC cells leads to mis-patterned facial skeleton and congenital bony syngnathia, and suggest an implication of mutations in BMP signaling pathway in human bony syngnathia.
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