ArticleProceedings of the National Academy of Sciences of the United States of America2001
Targeted mutagenesis of Lis1 disrupts cortical development and LIS1 homodimerization.
Article in Proceedings of the National Academy of Sciences of the United States of America, 2001. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 60 papers.
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Who cites it
60 citing papers in PubMed, 151 citations in OpenAlex.
- Adaptor-mediated recruitment of three dyneins to dynactin enhances force generation.Nature cell biology · 2026Article
- Selective Lis1 inactivation disrupts migration and positioning of cortical somatostatin interneurons.Scientific reports · 2026Article
- Article
- NUDC Is Critical for Mitosis and Postmitotic Cell Maintenance Through Its Modulation of Dynein and Actin Cytoskeletal Reorganization.Advances in experimental medicine and biology · 2025Review
- Using cortical organoids to understand the pathogenesis of malformations of cortical development.Frontiers in neuroscience · 2024Review
- LIS1 RNA-binding orchestrates the mechanosensitive properties of embryonic stem cells in AGO2-dependent and independent ways.Nature communications · 2023Article
- Review
- Properties of the epileptiform activity in the cingulate cortex of a mouse model of LIS1 dysfunction.Brain structure & function · 2022Article
- Structural Consequence of Non-Synonymous Single-Nucleotide Variants in the N-Terminal Domain of LIS1.International journal of molecular sciences · 2022Article
- Abnormalities in Cortical GABAergic Interneurons of the Primary Motor Cortex Caused byFrontiers in cell and developmental biology · 2022Article
- Roots of the Malformations of Cortical Development in the Cell Biology of Neural Progenitor Cells.Frontiers in neuroscience · 2021Review
- Interneuron Heterotopia in the Lis1 Mutant Mouse Cortex Underlies a Structural and Functional Schizophrenia-Like Phenotype.Frontiers in cell and developmental biology · 2021Article
- Interplay of LIS1 and MeCP2: Interactions and Implications With the Neurodevelopmental Disorders Lissencephaly and Rett Syndrome.Frontiers in cellular neuroscience · 2019Article
- WD40-repeat 47, a microtubule-associated protein, is essential for brain development and autophagy.Proceedings of the National Academy of Sciences of the United States of America · 2017Article
- INTERNEURONOPATHIES AND THEIR ROLE IN EARLY LIFE EPILEPSIES AND NEURODEVELOPMENTAL DISORDERS.Epilepsia open · 2017Article
- Developmental downregulation of LIS1 expression limits axonal extension and allows axon pruning.Biology open · 2017Article
- Fundamental Elements in Autism: From Neurogenesis and Neurite Growth to Synaptic Plasticity.Frontiers in cellular neuroscience · 2017Review
- 14-3-3 Proteins in Brain Development: Neurogenesis, Neuronal Migration and Neuromorphogenesis.Frontiers in molecular neuroscience · 2017Review
- Cross-Species Integrative Functional Genomics in GeneWeaver Reveals a Role for Pafah1b1 in Altered Response to Alcohol.Frontiers in behavioral neuroscience · 2016Article
- Cell-intrinsic drivers of dendrite morphogenesis.Development (Cambridge, England) · 2013Review
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Authors and funding
12 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Lissencephaly is a severe brain malformation in humans. To study the function of the gene mutated in lissencephaly (LIS1), we deleted the first coding exon from the mouse Lis1 gene. The deletion resulted in a shorter protein (sLIS1) that initiates from the second methionine, a unique situation because most LIS1 mutations result in a null allele. This mutation mimics a mutation described in one lissencephaly patient with a milder phenotype. Homozygotes are early lethal, although heterozygotes are viable and fertile. Most strikingly, the morphology of cortical neurons and radial glia is aberrant in the developing cortex, and the neurons migrate more slowly. This is the first demonstration, to our knowledge, of a cellular abnormality in the migrating neurons after Lis1 mutation. Moreover, cortical plate splitting and thalomocortical innervation are also abnormal. Biochemically, the mutant protein is not capable of dimerization, and enzymatic activity is elevated in the embryos, thus a demonstration of the in vivo role of LIS1 as a subunit of PAF-AH. This mutation allows us to determine a hierarchy of functions that are sensitive to LIS1 dosage, thus promoting our understanding of the role of LIS1 in the developing cortex.
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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.