ArticleProceedings of the National Academy of Sciences of the United States of America2014
prickle modulates microtubule polarity and axonal transport to ameliorate seizures in flies.
Article in Proceedings of the National Academy of Sciences of the United States of America, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 47 papers.
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Who cites it
47 citing papers in PubMed, 66 citations in OpenAlex.
- Strained actin binding by the Prickle2 LIM domains and their regulation in the full-length protein.Molecular biology of the cell · 2026Article
- Automated identification of ataxia and convulsions in hyperexcitablebioRxiv : the preprint server for biology · 2025Article
- Evolving Insights into Prickle2 in Neurodevelopment and Neurological Disorders.Molecular neurobiology · 2025Review
- Decoding the role of microtubules: a trafficking road for vesicle.Theranostics · 2025Review
- Automated identification of ataxia and convulsions in hyperexcitablemicroPublication biology · 2025Article
- Decoding Epilepsy: Prickle2 and Multifaceted Molecular Pathway Connections.Current pharmaceutical design · 2025Review
- Integrative analysis of blood transcriptome profiles in small-cell lung cancer patients for identification of novel chemotherapy resistance-related biomarkers.Frontiers in immunology · 2024Article
- Microtubule stabilising peptides: new paradigm towards management of neuronal disorders.RSC medicinal chemistry · 2023Review
- Downregulation of oxidative stress-mediated glial innate immune response suppresses seizures in a fly epilepsy model.Cell reports · 2023Article
- Epilepsy geneiScience · 2023Article
- The core PCP protein Prickle2 regulates axon number and AIS maturation by binding to AnkG and modulating microtubule bundling.Science advances · 2022Article
- Characterization ofmicroPublication biology · 2022Article
- Distinct overlapping functions for Prickle1 and Prickle2 in the polarization of the airway epithelium.Frontiers in cell and developmental biology · 2022Article
- A familiar study on self-limited childhood epilepsy patients using hIPSC-derived neurons shows a bias towards immaturity at the morphological, electrophysiological and gene expression levels.Stem cell research & therapy · 2021Article
- Fly seizure EEG: field potential activity in theJournal of neurogenetics · 2021Article
- The Ubiquitinated Axon: Local Control of Axon Development and Function by Ubiquitin.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2021Review
- How do the Fat-Dachsous and core planar polarity pathways act together and independently to coordinate polarized cell behaviours?Open biology · 2021Review
- The Cytoplasmic LIM Domain Protein Espinas Contributes to Photoreceptor Layer Selection in the Visual System.Biology · 2020Article
- Investigating Developmental and Epileptic Encephalopathy UsingInternational journal of molecular sciences · 2020Review
- Prickle2 and Igsf9b Coordinately Regulate the Cytoarchitecture of the Axon Initial Segment.Cell structure and function · 2020Article
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Authors and funding
11 authors at 7 institutions in 3 countries.
Funding
Abstract
Recent analyses in flies, mice, zebrafish, and humans showed that mutations in prickle orthologs result in epileptic phenotypes, although the mechanism responsible for generating the seizures was unknown. Here, we show that Prickle organizes microtubule polarity and affects their growth dynamics in axons of Drosophila neurons, which in turn influences both anterograde and retrograde vesicle transport. We also show that enhancement of the anterograde transport mechanism is the cause of the seizure phenotype in flies, which can be suppressed by reducing the level of either of two Kinesin motor proteins responsible for anterograde vesicle transport. Additionally, we show that seizure-prone prickle mutant flies have electrophysiological defects similar to other fly mutants used to study seizures, and that merely altering the balance of the two adult prickle isoforms in neurons can predispose flies to seizures. These data reveal a previously unidentified pathway in the pathophysiology of seizure disorders and provide evidence for a more generalized cellular mechanism whereby Prickle mediates polarity by influencing microtubule-mediated transport.
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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.