ArticleThe Journal of neuroscience : the official journal of the Society for Neuroscience2009
Selective cortical layering abnormalities and behavioral deficits in cortex-specific Pax6 knock-out mice.
Article in The Journal of neuroscience : the official journal of the Society for Neuroscience, 2009. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 62 papers.
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Who cites it
62 citing papers in PubMed, 112 citations in OpenAlex.
- Regeneration-Associated Factors in the Regulation of Adult and Post-Traumatic Neurogenesis in the Forebrain of Fish and Other Vertebrates.International journal of molecular sciences · 2025Review
- Mouse cortical cellular diversification through lineage progression of radial glia.Genes & development · 2025Article
- Article
- Transcriptional regulation in the development and dysfunction of neocortical projection neurons.Neural regeneration research · 2024Review
- H3 Acetylation-Induced Basal Progenitor Generation and Neocortex Expansion Depends on the Transcription Factor Pax6.Biology · 2024Article
- The role of prenatal choline and its impact on neurodevelopmental disorders.Frontiers in nutrition · 2024Review
- Co-administration of extracellular matrix-based biomaterials with neural stem cell transplantation for treatment of central nervous system injury.Frontiers in neuroscience · 2023Review
- Pax6 limits the competence of developing cerebral cortical cells to respond to inductive intercellular signals.PLoS biology · 2022Article
- Evolution of genetic mechanisms regulating cortical neurogenesis.Developmental neurobiology · 2022Review
- Thirty Years' History since the Discovery of Pax6: From Central Nervous System Development to Neurodevelopmental Disorders.International journal of molecular sciences · 2022Review
- BAF (mSWI/SNF) complex regulates mediolateral cortical patterning in the developing forebrain.Frontiers in cell and developmental biology · 2022Article
- Pax6 mutant cerebral organoids partially recapitulate phenotypes of Pax6 mutant mouse strains.PloS one · 2022Article
- H3 acetylation selectively promotes basal progenitor proliferation and neocortex expansion.Science advances · 2021Article
- Structural and Functional Aspects of the Neurodevelopmental GeneFrontiers in molecular neuroscience · 2021Review
- Conditional Loss of BAF (mSWI/SNF) Scaffolding Subunits Affects Specification and Proliferation of Oligodendrocyte Precursors in Developing Mouse Forebrain.Frontiers in cell and developmental biology · 2021Article
- Largazole is a Brain-Penetrant Class I HDAC Inhibitor with Extended Applicability to Glioblastoma and CNS Diseases.ACS chemical neuroscience · 2020Article
- Use of ocular biomarkers as a potential tool for early diagnosis of Alzheimer's disease.Indian journal of ophthalmology · 2020Review
- Global and age-related neuroanatomical abnormalities in a Pax6-deficient mouse model of aniridia suggests a role for Pax6 in adult structural neuroplasticity.Brain research · 2020Article
- Foxp1 Regulates Neural Stem Cell Self-Renewal and Bias Toward Deep Layer Cortical Fates.Cell reports · 2020Article
- RBM15 Modulates the Function of Chromatin Remodeling Factor BAF155 Through RNA Methylation in Developing Cortex.Molecular neurobiology · 2019Article
2 more citing papers are in PubMed but not listed here.
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Authors and funding
8 authors at 6 institutions in 4 countries.
Funding
Abstract
The transcription factor Pax6 has been implicated in neocortical neurogenesis in vertebrates, including humans. Analyses of the role of Pax6 in layer formation and cognitive abilities have been hampered by perinatal lethality of Pax6 mutants. Here, we generated viable mutants exhibiting timed, restricted inactivation of Pax6 during early and late cortical neurogenesis using Emx1-Cre and hGFAP-Cre lines, respectively. The disruption of Pax6 at the onset of neurogenesis using Emx1-Cre line resulted in premature cell cycle exit of early progenitors, increase of early born neuronal subsets located in the marginal zone and lower layers, and a nearly complete absence of upper layer neurons, especially in the rostral cortex. Furthermore, progenitors, which accumulated in the enlarged germinal neuroepithelium at the pallial/subpallial border in the Pax6 mutants, produced an excess of oligodendrocytes. The inactivation of Pax6 after generation of the lower neuronal layers using hGFAP-Cre line did not affect specification or numbers of late-born neurons, indicating that the severe reduction of upper layer neurons in Pax6 deficiency is mostly attributable to a depletion of the progenitor pool, available for late neurogenesis. We further show that Pax6(fl/fl);Emx1-Cre mutants exhibited deficiencies in sensorimotor information integration, and both hippocampus-dependent short-term and neocortex-dependent long-term memory recall. Because a majority of the morphological and behavior disabilities of the Pax6 mutant mice parallel abnormalities reported for aniridia patients, a condition caused by PAX6 haploinsufficiency, the Pax6 conditional mutant mice generated here represent a valuable genetic tool to understand how the developmental cortical disruption can lead to a human behavior abnormality.
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Registered trials
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.