ArticleEMBO reports2020
ECE2 regulates neurogenesis and neuronal migration during human cortical development.
Article in EMBO reports, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 36 papers, 1 of them a synthesis that pooled it.
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Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
36 citing papers in PubMed, 1 synthesis or guideline pooled it, 54 citations in OpenAlex.
- Mapping the Cerebral Organoid Landscape: A Systematic Review of Preclinical 3D Models in Neuroscience.Advanced healthcare materials · 2026Pooled it
- PACS1 syndrome variant alters the proteomic landscape of developing cortical organoids.iScience · 2026Article
- Article
- Gene variants in periventricular nodular heterotopia.Acta epileptologica · 2026Review
- WWP1 gain-of-function drives developmental anoikis through TGFβ pathway during neurodevelopment.Cell death discovery · 2026Article
- Shaping the Neocortex: Radial Glia and Astrocytes in Development and Evolution.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2025Review
- Exploring the evolutionary conservation and functional implications of Endothelin Converting Enzyme 1 (ECE1) in lamprey (Lethenteron reissneri).Fish physiology and biochemistry · 2025Article
- Human dorsal forebrain organoids show differentiation-state-specific protein secretion.iScience · 2025Article
- Modelling human brain development and disease with organoids.Nature reviews. Molecular cell biology · 2025Review
- Early spinal cord development: from neural tube formation to neurogenesis.Nature reviews. Neuroscience · 2025Review
- Mapping fatigue: discovering brain regions and genes linked to fatigue susceptibility.Journal of translational medicine · 2025Article
- Neuronal hyperactivity in neurons derived from individuals with gray matter heterotopia.Nature communications · 2025Article
- Differential susceptibility to prenatal stress exposure in serotonin transporter-deficient female mice-an epigenetic exploration.Frontiers in neuroscience · 2025Article
- Suppression of ferroptosis by vitamin A or radical-trapping antioxidants is essential for neuronal development.Nature communications · 2024Article
- Radial glia progenitor polarity in health and disease.Frontiers in cell and developmental biology · 2024Review
- Neurodevelopmental defects in human cortical organoids withScience advances · 2023Article
- Metformin rescues migratory deficits of cells derived from patients with periventricular heterotopia.EMBO molecular medicine · 2023Article
- Non-muscle myosins control radial glial basal endfeet to mediate interneuron organization.PLoS biology · 2023Article
- Modeling congenital brain malformations with brain organoids: a narrative review.Translational pediatrics · 2023Review
- The genetic architecture of phenotypic diversity in the Betta fish (Science advances · 2022Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
10 authors at 4 institutions in 3 countries.
Funding
Abstract
During embryonic development, excitatory projection neurons migrate in the cerebral cortex giving rise to organised layers. Periventricular heterotopia (PH) is a group of aetiologically heterogeneous disorders in which a subpopulation of newborn projection neurons fails to initiate their radial migration to the cortex, ultimately resulting in bands or nodules of grey matter lining the lateral ventricles. Although a number of genes have been implicated in its cause, currently they only satisfactorily explain the pathogenesis of the condition for 50% of patients. Novel gene discovery is complicated by the extreme genetic heterogeneity recently described to underlie its cause. Here, we study the neurodevelopmental role of endothelin-converting enzyme-2 (ECE2) for which two biallelic variants have been identified in two separate patients with PH. Our results show that manipulation of ECE2 levels in human cerebral organoids and in the developing mouse cortex leads to ectopic localisation of neural progenitors and neurons. We uncover the role of ECE2 in neurogenesis, and mechanistically, we identify its involvement in the generation and secretion of extracellular matrix proteins in addition to cytoskeleton and adhesion.
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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.