ArticleFrontiers in integrative neuroscience2021
Auditory Brain Stem Responses in the C57BL/6J Fragile X Syndrome-Knockout Mouse Model.
Article in Frontiers in integrative neuroscience, 2021. 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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14 citing papers in PubMed, 23 citations in OpenAlex.
- The effects of interaural time and level differences on the binaural interaction component of the auditory brainstem response in Cope's gray treefrogs.Journal of comparative physiology. A, Neuroethology, sensory, neural, and behavioral physiology · 2026Article
- 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
- Binaural Processing Deficits in Autism Spectrum Disorder.Audiology research · 2026Review
- Differences in auditory brainstem responses between laboratory-reared and wild-caught prairie voles (Microtus ochrogaster).PloS one · 2026Article
- Loss of Fmr1 reorganizes the multi-elemental composition across tissues in Fragile X Syndrome mice.PloS one · 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
- Cortical layer-specific abnormalities in auditory responses in a mouse model of Fragile X Syndrome.Neurobiology of disease · 2025Article
- Auditory brainstem responses in two closely related Peromyscus species (Peromyscus leucopus and Peromyscus maniculatus).The Journal of the Acoustical Society of America · 2025Article
- Sex-specific loss of mitochondrial membrane integrity in the auditory brainstem of a mouse model of Fragile X Syndrome.Open biology · 2025Article
- Hearing ability of prairie voles (Microtus ochrogaster).The Journal of the Acoustical Society of America · 2024Article
- 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
- Auditory brainstem development of naked mole-rats (Proceedings. Biological sciences · 2022Article
- Absence of the Fragile X messenger ribonucleoprotein alters response patterns to sounds in the auditory midbrain.Frontiers in neuroscience · 2022Article
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2 authors at 1 institution in 1 country.
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Abstract
Sensory hypersensitivity, especially in the auditory system, is a common symptom in Fragile X syndrome (FXS), the most common monogenic form of intellectual disability. However, linking phenotypes across genetic background strains of mouse models has been a challenge and could underly some of the issues with translatability of drug studies to the human condition. This study is the first to characterize the auditory brain stem response (ABR), a minimally invasive physiological readout of early auditory processing that is also used in humans, in a commonly used mouse background strain model of FXS, C57BL/6J. We measured morphological features of pinna and head and used ABR to measure the hearing range, and monaural and binaural auditory responses in hemizygous males, homozygous females, and heterozygous females compared with those in wild-type mice. Consistent with previous study, we showed no difference in morphological parameters across genotypes or sexes. There was no significant difference in hearing range between the sexes or genotypes, however there was a trend towards high frequency hearing loss in male FXS mice. In contrast, female mice with homozygous FXS had a decreased amplitude of wave IV of the monaural ABR, while there was no difference in males for amplitudes and no change in latency of ABR waveforms across sexes and genotypes. Finally, males with FXS had an increased latency of the binaural interaction component (BIC) at 0 interaural timing difference compared with that in wild-type males. These findings further clarify auditory brain stem processing in FXS by adding more information across genetic background strains allowing for a better understanding of shared phenotypes.
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