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- W4387492756 abstract "Attention deficit is one of the most prominent and disabling symptoms in Fragile X Syndrome (FXS). Hypersensitivity to sensory stimuli contributes to attention difficulties by overwhelming and/or distracting affected individuals, which disrupts activities of daily living at home and learning at school. We find that auditory or visual distractors selectively impair visual discrimination performance in humans and mice with FXS, but not their typically developing controls. In both species, males and females were examined. Vasoactive intestinal polypeptide (VIP) neurons were significantly modulated by incorrect responses in the post-stimulus period during early distractor trials in WT mice, consistent with their known role as 'error' signals. Strikingly, however, VIP cells from Fmr1-/- mice showed little modulation in error trials, and this correlated with their poor performance on the distractor task. Thus, VIP interneurons and their reduced modulatory influence on pyramidal cells, could be a potential therapeutic target for attentional difficulties in FXS.Significance StatementSensory hypersensitivity, impulsivity, and persistent inattention are among the most consistent clinical features of FXS, all of which impede daily functioning and create barriers to learning. However, the neural mechanisms underlying sensory over-reactivity remain elusive. To overcome a significant challenge in translational FXS research we demonstrate a compelling alignment of sensory over-reactivity in both humans with FXS and Fmr1-/- mice (the principal animal model of FXS) using a novel analogous distractor task. Two photon microscopy in mice reveal that dysfunctional VIP cells contribute to susceptibility to distractors. Implementing research efforts we describe here, can help identify dysfunctional neural mechanisms associated not only with sensory issues, but broader impairments, including those in learning and cognition." @default.
- W4387492756 created "2023-10-11" @default.
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- W4387492756 date "2023-10-10" @default.
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- W4387492756 title "Hypersensitivity to distractors in Fragile X syndrome from loss of modulation of cortical VIP interneurons" @default.
- W4387492756 doi "https://doi.org/10.1523/jneurosci.0571-23.2023" @default.
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