Early developmental characterization of gap-induced prepulse inhibition and habituation of auditory startle response in a Fragile X Syndrome mouse model.
Aug 2026· Developmental Neuroscience· pp.
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· 0 citations
Medicine
TL;DR
The findings suggest that behavioral responses to auditory stimuli are dynamic during development and differ between females and males, which could be an ideal window for addressing auditory hypersensitivity in FXS.
It is concluded that CA1 PCs hyperexcitability from Frm1 KO mice was detectable at late postnatal development (P21-28), but not earlier (P8-15), and was due to a shifting of action potential voltage threshold to more hyperpolarized values.
M. A. Luque, L. Dufresne, B. Torres et al.· Neuroscience· 0 citations
Prepulse inhibition (PPI) of the acoustic startle reflex is a widely used operational measure of sensorimotor gating that is disrupted in several neuropsychiatric disorders. PPI deficits are a hallmark feature of schizophrenia, often associated with attentional and cognitive impairments. Although PPI is extensively use...
L. Martinetti, Erika Correll, A. E. Cuadra et al.· Journal of Neuroscience· 0 citations
A causal link from FMRP loss to circuit dysfunction and ERK pathway dysregulation is established, and it is suggested that targeting this pathway may offer a promising strategy for treating social dysfunction in ASD and FXS.
It is shown that early auditory dysfunction is a conserved consequence of Cntnap2 loss early in development but normalized in adulthood, similar to the Cntnap2 knockout rat model, and reinforces the need to consider sex as a biological variable when investigating neural mechanisms underlying neurodevelopmental disorder...
N. Chan, S. D. I. Seheult, A. Warr et al.· bioRxiv· 0 citations
Metabotropic glutamate receptors (mGluR) are G-protein coupled receptors that regulate neuroplasticity and have been shown to play a role in auditory processing. However, not much is known about the expression of mGluRs during cochlear nucleus development in the context of Fragile X syndrome (FXS). FXS is the most comm...
H. Sakano, Jason W. Wheeler, Justine Lam et al.· bioRxiv· 0 citations
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