ArticlePloS one2015
Deletion of Fmr1 alters function and synaptic inputs in the auditory brainstem.
Article in PloS one, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 53 papers, 2 of them syntheses that pooled it.
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Who cites it
53 citing papers in PubMed, 2 syntheses or guidelines pooled it, 73 citations in OpenAlex.
- Auditory processing in rodent models of autism: a systematic review.Journal of neurodevelopmental disorders · 2022Pooled it
- A Systematic Review of Brainstem Contributions to Autism Spectrum Disorder.Frontiers in integrative neuroscience · 2021Pooled it
- Computational Model for Synthesizing Auditory Brainstem Responses to Assess Neuronal Alterations in Aging and Autistic Animal Models.Journal of the Association for Research in Otolaryngology : JARO · 2026Article
- Altered Auditory Maturation in Fragile X Syndrome and Its Involvement in Audiogenic Seizure Susceptibility.Autism research : official journal of the International Society for Autism Research · 2026Article
- Sex-specific loss of mitochondrial membrane integrity in the auditory brainstem of a mouse model of Fragile X Syndrome.Open biology · 2025Article
- Delayed structural maturation of inner hair cell ribbon synapses in a mouse model of fragile X syndrome.Frontiers in molecular neuroscience · 2025Article
- Specialization of the brain for language in children with Fragile X Syndrome: a functional Near Infrared Spectroscopy study.Journal of neurodevelopmental disorders · 2024Article
- Stem Cell Secretions as a Potential Therapeutic Agent for Autism Spectrum Disorder: A Narrative Review.Stem cell reviews and reports · 2024Review
- A novel combination treatment for fragile X syndrome predicted using computational methods.Brain communications · 2024Article
- Hyperacusis in the AdultInternational journal of molecular sciences · 2023Article
- Current Best Practices for Analysis of Dendritic Spine Morphology and Number in Neurodevelopmental Disorder Research.ACS chemical neuroscience · 2023Review
- Axonal and presynaptic FMRP: Localization, signal, and functional implications.Hearing research · 2023Review
- Peripheral Fragile X messenger ribonucleoprotein is required for the timely closure of a critical period for neuronal susceptibility in the ventral cochlear nucleus.Frontiers in cellular neuroscience · 2023Article
- Cellular distribution of the Fragile X mental retardation protein in the inner ear: a developmental and comparative study in the mouse, rat, gerbil, and chicken.The Journal of comparative neurology · 2023Article
- Tonotopic differentiation of presynaptic neurotransmitter-releasing machinery in the auditory brainstem during the prehearing period and its selective deficits in Fmr1 knockout mice.The Journal of comparative neurology · 2022Article
- Peripheral Auditory Nerve Impairment in a Mouse Model of Syndromic Autism.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2022Article
- Largely unaffected auditory and visual sensory processing phenotypes in the evoked potentials of Fmr1 KO2 mice.The European journal of neuroscience · 2022Article
- A sound-driven cortical phase-locking change in the Fmr1 KO mouse requires Fmr1 deletion in a subpopulation of brainstem neurons.Neurobiology of disease · 2022Article
- Spontaneous seizures in adult Fmr1 knockout mice: FVB.129P2-Pde6bEpilepsy research · 2022Article
- ReducedFrontiers in cell and developmental biology · 2022Article
Corrections and comments
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Authors and funding
3 authors at 1 institution in 1 country.
Funding
Abstract
Fragile X Syndrome (FXS), a neurodevelopmental disorder, is the most prevalent single-gene cause of autism spectrum disorder. Autism has been associated with impaired auditory processing, abnormalities in the auditory brainstem response (ABR), and reduced cell number and size in the auditory brainstem nuclei. FXS is characterized by elevated cortical responses to sound stimuli, with some evidence for aberrant ABRs. Here, we assessed ABRs and auditory brainstem anatomy in Fmr1-/- mice, an animal model of FXS. We found that Fmr1-/- mice showed elevated response thresholds to both click and tone stimuli. Amplitudes of ABR responses were reduced in Fmr1-/- mice for early peaks of the ABR. The growth of the peak I response with sound intensity was less steep in mutants that in wild type mice. In contrast, amplitudes and response growth in peaks IV and V did not differ between these groups. We did not observe differences in peak latencies or in interpeak latencies. Cell size was reduced in Fmr1-/- mice in the ventral cochlear nucleus (VCN) and in the medial nucleus of the trapezoid body (MNTB). We quantified levels of inhibitory and excitatory synaptic inputs in these nuclei using markers for presynaptic proteins. We measured VGAT and VGLUT immunolabeling in VCN, MNTB, and the lateral superior olive (LSO). VGAT expression in MNTB was significantly greater in the Fmr1-/- mouse than in wild type mice. Together, these observations demonstrate that FXS affects peripheral and central aspects of hearing and alters the balance of excitation and inhibition in the auditory brainstem.
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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.