ArticleProceedings of the National Academy of Sciences of the United States of America2007
Cortical dysplasia and skull defects in mice with a Foxc1 allele reveal the role of meningeal differentiation in regulating cortical development.
Article in Proceedings of the National Academy of Sciences of the United States of America, 2007. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 63 papers.
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63 citing papers in PubMed.
- A transcriptional atlas of gyrification reveals dynamic spatio-temporal gene expression in the developing ferret cortex.Brain structure & function · 2026Article
- Duropathies: A Narrative Overview of a Neglected Concept-Part One: Anatomical, Embryological, and Pathophysiological Elements.NeuroSci · 2025Review
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- Meningeal-derived retinoic acid regulates neurogenesis via suppression of Notch and Sox2.Cell reports · 2025Article
- Perivascular NOTCH3+ Stem Cells Drive Meningioma Tumorigenesis and Resistance to Radiotherapy.Cancer discovery · 2024Article
- Functional analysis and transcriptome profile of meninges and skin fibroblasts from human-aged donors.Cell proliferation · 2024Article
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- Loss of Twist1 and balanced retinoic acid signaling from the meninges causes cortical folding in mice.Development (Cambridge, England) · 2023Article
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- Cranium growth, patterning and homeostasis.Development (Cambridge, England) · 2022Article
- Severe cerebellar malformations in mutant mice demonstrate a role for PDGF-C/PDGFRα signalling in cerebellar development.Biology open · 2022Article
- Regional brain development in fetuses with Dandy-Walker malformation: A volumetric fetal brain magnetic resonance imaging study.PloS one · 2022Article
- Characterization, isolation, and in vitro culture of leptomeningeal fibroblasts.Journal of neuroimmunology · 2021Article
- Central Nervous System Fibroblast-Like Cells in Stroke and Other Neurological Disorders.Stroke · 2021Review
- Meningiomas from a developmental perspective: exploring the crossroads between meningeal embryology and tumorigenesis.Acta neurochirurgica · 2021Review
- Single-Cell Transcriptomic Analyses of the Developing Meninges Reveal Meningeal Fibroblast Diversity and Function.Developmental cell · 2020Article
- Article
- Developmental biology of the meninges.Genesis (New York, N.Y. : 2000) · 2019Review
- Wnt/β-catenin signaling in the mouse embryonic cranial mesenchyme is required to sustain the emerging differentiated meningeal layers.Genesis (New York, N.Y. : 2000) · 2019Article
- Role of FOXC1 in regulating APSCs self-renewal via STI-1/PrPTheranostics · 2019Article
3 more citing papers are in PubMed but not listed here.
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6 authors.
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Abstract
We report the identification of a hypomorphic mouse allele for Foxc1 (Foxc1(hith)) that survives into adulthood revealing previously unknown roles for Foxc1 in development of the skull and cerebral cortex. This line of mice was recovered in a forward genetic screen using ENU mutagenesis to identify mutants with cortical defects. In the hith allele a missense mutation substitutes a Leu for a conserved Phe at amino acid 107, leading to destabilization of the protein without substantially altering transcriptional activity. Embryonic and postnatal histological analyses indicate that diminished Foxc1 protein expression in all three layers of meningeal cells in Foxc1(hith/hith) mice contributes to the cortical and skull defects in mutant mice and that the prominent phenotypes appear as the meninges differentiate into pia, arachnoid, and dura. Careful analysis of the cortical phenotypes shows that Foxc1(hith/hith) mice display detachment of radial glial endfeet, marginal zone heterotopias, and cortical dyslamination. These abnormalities have some features resembling defects in type 2 (cobblestone) lissencephaly or congenital muscular dystrophies but appear later in corticogenesis because of the delay in breakdown of the basement membrane. Our data reveal that the meninges regulate the development of the skull and cerebral cortex by controlling aspects of the formation of these neighboring structures. Furthermore, we provide evidence that defects in meningeal differentiation can lead to severe cortical dysplasia.
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