ArticleNeural development2016
Fragile X mental retardation protein knockdown in the developing Xenopus tadpole optic tectum results in enhanced feedforward inhibition and behavioral deficits.
Article in Neural development, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers.
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Who cites it
18 citing papers in PubMed, 28 citations in OpenAlex.
- Assessing circuit function in the developingFrontiers in behavioral neuroscience · 2026Review
- Accelerated protein retention expansion microscopy using microwave radiation.Cell reports methods · 2024Article
- X-Tracker: Automated Analysis of Xenopus Tadpole Visual Avoidance Behavior.bioRxiv : the preprint server for biology · 2024Article
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- Accelerated protein retention expansion microscopy using microwave radiation.bioRxiv : the preprint server for biology · 2024Article
- Strategies for dissecting the complexity of neurodevelopmental disorders.Trends in genetics : TIG · 2024Review
- Characterization of NaLife science alliance · 2024Article
- Identification and functional evaluation of GRIA1 missense and truncation variants in individuals with ID: An emerging neurodevelopmental syndrome.American journal of human genetics · 2022Article
- Role of matrix metalloproteinase-9 in neurodevelopmental deficits and experience-dependent plasticity ineLife · 2021Article
- Early Developmental Exposure to Fluoxetine and Citalopram Results in Different Neurodevelopmental Outcomes.Neuroscience · 2021Article
- Xenopus leads the way: Frogs as a pioneering model to understand the human brain.Genesis (New York, N.Y. : 2000) · 2021Review
- Experience-Dependent Development of Dendritic Arbors in Mouse Visual Cortex.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2020Article
- Presenilin Regulates Retinotectal Synapse Formation through EphB2 Receptor Processing.Developmental neurobiology · 2018Article
- Neural architecture: from cells to circuits.Journal of neurophysiology · 2018Review
- Modeling Fragile X Syndrome inFrontiers in molecular neuroscience · 2018Review
- Of Men and Mice: Modeling the Fragile X Syndrome.Frontiers in molecular neuroscience · 2018Article
- Neural Circuits: Reduced Inhibition in Fragile X Syndrome.Current biology : CB · 2017Article
- Article
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Authors and funding
8 authors at 3 institutions in 1 country.
Funding
Abstract
backgroundFragile X Syndrome is the leading monogenetic cause of autism and most common form of intellectual disability. Previous studies have implicated changes in dendritic spine architecture as the primary result of loss of Fragile X Mental Retardation Protein (FMRP), but recent work has shown that neural proliferation is decreased and cell death is increased with either loss of FMRP or overexpression of FMRP. The purpose of this study was to investigate the effects of loss of FMRP on behavior and cellular activity.
methodsWe knocked down FMRP expression using morpholino oligos in the optic tectum of Xenopus laevis tadpoles and performed a series of behavioral and electrophysiological assays. We investigated visually guided collision avoidance, schooling, and seizure propensity. Using single cell electrophysiology, we assessed intrinsic excitability and synaptic connectivity of tectal neurons.
resultsWe found that FMRP knockdown results in decreased swimming speed, reduced schooling behavior and decreased seizure severity. In single cells, we found increased inhibition relative to excitation in response to sensory input.
conclusionsOur results indicate that the electrophysiological development of single cells in the absence of FMRP is largely unaffected despite the large neural proliferation defect. The changes in behavior are consistent with an increase in inhibition, which could be due to either changes in cell number or altered inhibitory drive, and indicate that FMRP can play a significant role in neural development much earlier than previously thought.
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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.