ArticleThe Journal of biological chemistry2011
Duplicated gephyrin genes showing distinct tissue distribution and alternative splicing patterns mediate molybdenum cofactor biosynthesis, glycine receptor clustering, and escape behavior in zebrafish.
Article in The Journal of biological chemistry, 2011. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers.
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Who cites it
14 citing papers in PubMed, 20 citations in OpenAlex.
- Zebrafish oligodendrocyte lineage cells use the postsynaptic protein Gephyrin to myelinate GABAergic axons and limit myelin sheath growth.Nature communications · 2026Article
- Oligodendrocytes use postsynaptic proteins to coordinate myelin formation on axons of distinct neurotransmitter classes.bioRxiv : the preprint server for biology · 2024Article
- A zebrafish gephyrinb mutant distinguishes synaptic and enzymatic functions of Gephyrin.Neural development · 2024Article
- Phosphorylation of Gephyrin in Zebrafish Mauthner Cells Governs Glycine Receptor Clustering and Behavioral Desensitization to Sound.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2019Article
- Gephyrin suppresses lung squamous cell carcinoma development by reducing mTOR pathway activation.Cancer management and research · 2019Article
- Expansion microscopy of zebrafish for neuroscience and developmental biology studies.Proceedings of the National Academy of Sciences of the United States of America · 2017Article
- Sleep-Dependent Structural Synaptic Plasticity of Inhibitory Synapses in the Dendrites of Hypocretin/Orexin Neurons.Molecular neurobiology · 2017Article
- Spatial patterning of excitatory and inhibitory neuropil territories during spinal circuit development.The Journal of comparative neurology · 2017Article
- Gephyrin: a master regulator of neuronal function?Nature reviews. Neuroscience · 2014Review
- Distinct phenotypes in zebrafish models of human startle disease.Neurobiology of disease · 2013Article
- ZC4H2 mutations are associated with arthrogryposis multiplex congenita and intellectual disability through impairment of central and peripheral synaptic plasticity.American journal of human genetics · 2013Article
- Glycine receptors support excitatory neurotransmitter release in developing mouse visual cortex.The Journal of physiology · 2012Article
- Connexin 39.9 protein is necessary for coordinated activation of slow-twitch muscle and normal behavior in zebrafish.The Journal of biological chemistry · 2012Article
- Gephyrin, the enigmatic organizer at GABAergic synapses.Frontiers in cellular neuroscience · 2012Article
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Authors and funding
6 authors at 2 institutions in 2 countries.
Funding
Abstract
Gephyrin mediates the postsynaptic clustering of glycine receptors (GlyRs) and GABA(A) receptors at inhibitory synapses and molybdenum-dependent enzyme (molybdoenzyme) activity in non-neuronal tissues. Gephyrin knock-out mice show a phenotype resembling both defective glycinergic transmission and molybdenum cofactor (Moco) deficiency and die within 1 day of birth due to starvation and dyspnea resulting from deficits in motor and respiratory networks, respectively. To address whether gephyrin function is conserved among vertebrates and whether gephyrin deficiency affects molybdoenzyme activity and motor development, we cloned and characterized zebrafish gephyrin genes. We report here that zebrafish have two gephyrin genes, gphna and gphnb. The former is expressed in all tissues and has both C3 and C4 cassette exons, and the latter is expressed predominantly in the brain and spinal cord and harbors only C4 cassette exons. We confirmed that all of the gphna and gphnb splicing isoforms have Moco synthetic activity. Antisense morpholino knockdown of either gphna or gphnb alone did not disturb synaptic clusters of GlyRs in the spinal cord and did not affect touch-evoked escape behaviors. However, on knockdown of both gphna and gphnb, embryos showed impairments in GlyR clustering in the spinal cord and, as a consequence, demonstrated touch-evoked startle response behavior by contracting antagonistic muscles simultaneously, instead of displaying early coiling and late swimming behaviors, which are executed by side-to-side muscle contractions. These data indicate that duplicated gephyrin genes mediate Moco biosynthesis and control postsynaptic clustering of GlyRs, thereby mediating key escape behaviors in zebrafish.
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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.