ArticleThe Journal of neuroscience : the official journal of the Society for Neuroscience2013
α2-Chimaerin regulates a key axon guidance transition during development of the oculomotor projection.
Article in The Journal of neuroscience : the official journal of the Society for Neuroscience, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 26 papers.
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Who cites it
26 citing papers in PubMed, 49 citations in OpenAlex.
- Transcriptional profiling of extraocular motor neurons revealsbioRxiv : the preprint server for biology · 2026Article
- Birthdate aligns vestibular sensory neurons with central and motor partners across a sensorimotor reflex circuit for gaze stabilization.Development (Cambridge, England) · 2026Article
- Oculomotor axons use external and autocrine Slit signals for fasciculation, navigation, branching, and connectivity in mouse embryos.Research square · 2025Article
- Gene Identification for Ocular Congenital Cranial Motor Neuron Disorders Using Human Sequencing, Zebrafish Screening, and Protein Binding Microarrays.Investigative ophthalmology & visual science · 2025Article
- Motor neurons are dispensable for the assembly of a sensorimotor circuit for gaze stabilization.eLife · 2024Article
- Evolution and development of extraocular motor neurons, nerves and muscles in vertebrates.Annals of anatomy = Anatomischer Anzeiger : official organ of the Anatomische Gesellschaft · 2024Review
- Motor neurons are dispensable for the assembly of a sensorimotor circuit for gaze stabilization.bioRxiv : the preprint server for biology · 2024Article
- Two novel CHN1 variants identified in Duane retraction syndrome pedigrees disrupt development of ocular motor nerves in zebrafish.Journal of human genetics · 2024Article
- Neuronal birthdate reveals topography in a vestibular brainstem circuit for gaze stabilization.Current biology : CB · 2023Article
- Axonal Growth Abnormalities Underlying Ocular Cranial Nerve Disorders.Annual review of vision science · 2021Review
- The Rac-GAP alpha2-Chimaerin Signals via CRMP2 and Stathmins in the Development of the Ocular Motor System.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2021Article
- Oculomotor nerve guidance and terminal branching requires interactions with differentiating extraocular muscles.Developmental biology · 2021Article
- CHN1 promotes epithelial-mesenchymal transition via the Akt/GSK-3β/Snail pathway in cervical carcinoma.Journal of translational medicine · 2021Article
- Lysosomal Function and Axon Guidance: Is There a Meaningful Liaison?Biomolecules · 2021Review
- Article
- Cadherins regulate nuclear topography and function of developing ocular motor circuitry.eLife · 2020Article
- Isolation and Culture of Oculomotor, Trochlear, and Spinal Motor Neurons from Prenatal Islmn:GFP Transgenic Mice.Journal of visualized experiments : JoVE · 2019Article
- Ex Vivo Oculomotor Slice Culture from Embryonic GFP-Expressing Mice for Time-Lapse Imaging of Oculomotor Nerve Outgrowth.Journal of visualized experiments : JoVE · 2019Article
- Spinal RacGAP α-Chimaerin Is Required to Establish the Midline Barrier for Proper Corticospinal Axon Guidance.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2017Article
- Ocular congenital cranial dysinnervation disorders (CCDDs): insights into axon growth and guidance.Human molecular genetics · 2017Review
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Authors and funding
4 authors at 1 institution in 1 country.
Funding
Abstract
The ocular motor system consists of three nerves which innervate six muscles to control eye movements. In humans, defective development of this system leads to eye movement disorders, such as Duane Retraction Syndrome, which can result from mutations in the α2-chimaerin signaling molecule. We have used the zebrafish to model the role of α2-chimaerin during development of the ocular motor system. We first mapped ocular motor spatiotemporal development, which occurs between 24 and 72 h postfertilization (hpf), with the oculomotor nerve following an invariant sequence of growth and branching to its muscle targets. We identified 52 hpf as a key axon guidance "transition," when oculomotor axons reach the orbit and select their muscle targets. Live imaging and quantitation showed that, at 52 hpf, axons undergo a switch in behavior, with striking changes in the dynamics of filopodia. We tested the role of α2-chimaerin in this guidance process and found that axons expressing gain-of-function α2-chimaerin isoforms failed to undergo the 52 hpf transition in filopodial dynamics, leading to axon stalling. α2-chimaerin loss of function led to ecotopic and misguided branching and hypoplasia of oculomotor axons; embryos had defective eye movements as measured by the optokinetic reflex. Manipulation of chimaerin signaling in oculomotor neurons in vitro led to changes in microtubule stability. These findings demonstrate that a correct level of α2-chimaerin signaling is required for key oculomotor axon guidance decisions, and provide a zebrafish model for Duane Retraction Syndrome.
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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.