ArticleScientific reports2016
Sensory hypo-excitability in a rat model of fetal development in Fragile X Syndrome.
Article in Scientific reports, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 26 papers.
What it found
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
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Who cites it
26 citing papers in PubMed, 38 citations in OpenAlex.
- Compensatory Regulation of Excitation/Inhibition Balance in the Ventral Hippocampus: Insights from Fragile X Syndrome.Biology · 2025Review
- Peripheral contributions to resting state brain dynamics.Nature communications · 2024Article
- Purinergic Signalling Mediates Aberrant Excitability of Developing Neuronal Circuits in the Fmr1 Knockout Mouse Model.Molecular neurobiology · 2024Article
- A working taxonomy for describing the sensory differences of autism.Molecular autism · 2023Review
- Mouse models of fragile X-related disorders.Disease models & mechanisms · 2023Review
- Largely unaffected auditory and visual sensory processing phenotypes in the evoked potentials of Fmr1 KO2 mice.The European journal of neuroscience · 2022Article
- 5-HT5A Receptor Antagonist ASP5736 Ameliorates Several Abnormal Behaviors in an Fmr1-Targeted Transgenic Male Rat Model of Fragile X Syndrome.The international journal of neuropsychopharmacology · 2022Article
- Mechanisms Driving the Emergence of Neuronal Hyperexcitability in Fragile X Syndrome.International journal of molecular sciences · 2022Review
- Restoration of FMRP expression in adult V1 neurons rescues visual deficits in a mouse model of fragile X syndrome.Protein & cell · 2022Article
- Cortical interneurons in autism.Nature neuroscience · 2021Review
- Review
- Gamma power abnormalities in a Fmr1-targeted transgenic rat model of fragile X syndrome.Scientific reports · 2020Article
- Decreased reproducibility and abnormal experience-dependent plasticity of network dynamics in Fragile X circuits.Scientific reports · 2020Article
- Rat models of human diseases and related phenotypes: a systematic inventory of the causative genes.Journal of biomedical science · 2020Review
- Cortical neurons derived from human pluripotent stem cells lacking FMRP display altered spontaneous firing patterns.Molecular autism · 2020Article
- Increased Excitation-Inhibition Ratio Stabilizes Synapse and Circuit Excitability in Four Autism Mouse Models.Neuron · 2019Article
- Homeostatic Intrinsic Plasticity Is Functionally Altered in Fmr1 KO Cortical Neurons.Cell reports · 2019Article
- The Application of Adeno-Associated Viral Vector Gene Therapy to the Treatment of Fragile X Syndrome.Brain sciences · 2019Review
- Article
- Thalamocortical function in developing sensory circuits.Current opinion in neurobiology · 2018Review
Corrections and comments
- Erratum issued
Authors and funding
4 authors at 1 institution in 1 country.
Funding
Abstract
Fragile X syndrome (FXS) is characterized by sensory hyper-sensitivity, and animal models suggest that neuronal hyper-excitability contributes to this phenotype. To understand how sensory dysfunction develops in FXS, we used the rat model (FMR-KO) to quantify the maturation of cortical visual responses from the onset of responsiveness prior to eye-opening, through age equivalents of human juveniles. Rather than hyper-excitability, visual responses before eye-opening had reduced spike rates and an absence of early gamma oscillations, a marker for normal thalamic function at this age. Despite early hypo-excitability, the developmental trajectory of visual responses in FMR-KO rats was normal, and showed the expected loss of visually evoked bursting at the same age as wild-type, two days before eye-opening. At later ages, during the third and fourth post-natal weeks, signs of mild hyper-excitability emerged. These included an increase in the visually-evoked firing of regular spiking, presumptive excitatory, neurons, and a reduced firing of fast-spiking, presumptive inhibitory, neurons. Our results show that early network changes in the FMR-KO rat arise at ages equivalent to fetal humans and have consequences for excitability that are opposite those found in adults. This suggests identification and treatment should begin early, and be tailored in an age-appropriate manner.
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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.