ArticleThe Journal of clinical investigation2012
p38 Inhibition ameliorates skin and skull abnormalities in Fgfr2 Beare-Stevenson mice.
Article in The Journal of clinical investigation, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 32 papers.
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Who cites it
32 citing papers in PubMed.
- Temporally regulated FGFR2-retinoic acid signaling mediates dura mater-suture mesenchyme interactions to prevent craniosynostosis in mice.International journal of oral science · 2026Article
- The role of Pth1r in posterior cranium cartilage regulation and craniosynostosis.Nature communications · 2026Article
- P38α MAPK-induced senescence in cranial suture progenitor cells promotes craniosynostosis.Communications biology · 2025Article
- Nf2-FAK signaling axis is critical for cranial bone ossification and regeneration.Nature communications · 2025Article
- Targeted allele-specificMolecular therapy. Nucleic acids · 2025Article
- The CMS19 disease model specifies a pivotal role for collagen XIII in bone homeostasis.Scientific reports · 2022Article
- Article
- p38-MAPK-mediated translation regulation during early blastocyst development is required for primitive endoderm differentiation in mice.Communications biology · 2021Article
- Genotype-Phenotype Correlation of Tracheal Cartilaginous Sleeves and Fgfr2 Mutations in Mice.The Laryngoscope · 2021Article
- 2b or Not 2b: How Opposing FGF Receptor Splice Variants Are Blocking Progress in Precision Oncology.Journal of oncology · 2021Review
- RUNX2-modifying enzymes: therapeutic targets for bone diseases.Experimental & molecular medicine · 2020Review
- Article
- Article
- Report of a Father With Congenital Bilateral Absence of the Vas Deferens Fathering a Child With Beare-Stevenson Syndrome.Frontiers in genetics · 2020Article
- Current Approaches in the Development of Molecular and Pharmacological Therapies in Craniosynostosis Utilizing Animal Models.Molecular syndromology · 2019Review
- Mouse Models of Syndromic Craniosynostosis.Molecular syndromology · 2019Review
- Midface and upper airway dysgenesis in FGFR2-related craniosynostosis involves multiple tissue-specific and cell cycle effects.Development (Cambridge, England) · 2018Article
- Atypical Skin Manifestations inMolecular syndromology · 2018Article
- Genetic advances in craniosynostosis.American journal of medical genetics. Part A · 2017Review
- Tracheal cartilaginous sleeves in children with syndromic craniosynostosis.Genetics in medicine : official journal of the American College of Medical Genetics · 2017Article
Corrections and comments
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Authors and funding
15 authors.
Funding
Abstract
Beare-Stevenson cutis gyrata syndrome (BSS) is a human genetic disorder characterized by skin and skull abnormalities. BSS is caused by mutations in the FGF receptor 2 (FGFR2), but the molecular mechanisms that induce skin and skull abnormalities are unclear. We developed a mouse model of BSS harboring a FGFR2 Y394C mutation and identified p38 MAPK as an important signaling pathway mediating these abnormalities. Fgfr2+/Y394C mice exhibited epidermal hyperplasia and premature closure of cranial sutures (craniosynostosis) due to abnormal cell proliferation and differentiation. We found ligand-independent phosphorylation of FGFR2 and activation of p38 signaling in mutant skin and calvarial tissues. Treating Fgfr2+/Y394C mice with a p38 kinase inhibitor attenuated skin abnormalities by reversing cell proliferation and differentiation to near normal levels. This study reveals the pleiotropic effects of the FGFR2 Y394C mutation evidenced by cutis gyrata, acanthosis nigricans, and craniosynostosis and provides a useful model for investigating the molecular mechanisms of skin and skull development. The demonstration of a pathogenic role for p38 activation may lead to the development of therapeutic strategies for BSS and related conditions, such as acanthosis nigricans or craniosynostosis.
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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.