FX ENTRAIN: scientific context, study design, and biomarker driven brain-computer interfaces in neurodevelopmental conditions

Fragile X Syndrome (FXS), caused by the loss of function of the <i>Fmr1</i> gene, is characterized by varying degrees of intellectual disability, autistic features, and sensory hypersensitivity. Despite phenotypic rescue in animal deletion models, clinical trials in humans have been unsuccessful, likely due to the heterogeneous nature of FXS. To uncover the basis of individual- and subgroup-level variation driving treatment failures, we propose to test and modulate thalamocortical drive as a novel "bottom-up" neural probe to understand the mechanics of FXS-relevant circuits. Our study employs trial-level EEG analyses (neurodynamics) to detect fine-grained differences in brain activity using sensory and statistical learning paradigms in children with FXS, autism spectrum disorder (ASD), and typically developing controls. Parallel analysis in the FXS knockout mouse model will clarify its relevance to human FXS subgroups. In a randomized crossover study, we will evaluate the efficacy of closed-loop auditory entrainment, indexed on individual neurodynamic measures, aiming to normalize neural responses and enhance statistical learning performance. We anticipate this approach will yield opportunities to identify more effective early interventions that alter the trajectory of intellectual development in FXS.

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Publication Details

Journal
Frontiers in Neuroscience
Published
2025-09-09
DOI
https://doi.org/10.3389/fnins.2025.1618804
Citations
1
Primary Topic
Genetics and Neurodevelopmental Disorders
Type
article
Field-Weighted Citation Impact
1.82

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article

FX ENTRAIN: scientific context, study design, and biomarker driven brain-computer interfaces in neurodevelopmental conditions

Khaleel A. Razak, Elizabeth Blank, Donald L. Gilbert, Paul S. Horn et al.
1 citations
Frontiers in Neuroscience
Genetics and Neurodevelopmental Disorders
1.82
article

FX ENTRAIN: scientific context, study design, and biomarker driven brain-computer interfaces in neurodevelopmental conditions

Khaleel A. Razak, Elizabeth Blank, Donald L. Gilbert, Paul S. Horn, Devin K. Binder, Craig A. Erickson, L.D. Voorhees, Ernest V. Pedapati, Laura Batterink, Grace Westerkamp, Lauren E. Ethridge, Stephanie R. Jones, Meredith Nelson, Makoto Miyakoshi, Steve W. Wu, Lisa A. De Stefano, Elizabeth G. Smith, Debra L. Reisinger, Yanchen Liu, Peyton Siekierski, Jae Citarella
article en
1 citations

Abstract

Fragile X Syndrome (FXS), caused by the loss of function of the <i>Fmr1</i> gene, is characterized by varying degrees of intellectual disability, autistic features, and sensory hypersensitivity. Despite phenotypic rescue in animal deletion models, clinical trials in humans have been unsuccessful, likely due to the heterogeneous nature of FXS. To uncover the basis of individual- and subgroup-level variation driving treatment failures, we propose to test and modulate thalamocortical drive as a novel "bottom-up" neural probe to understand the mechanics of FXS-relevant circuits. Our study employs trial-level EEG analyses (neurodynamics) to detect fine-grained differences in brain activity using sensory and statistical learning paradigms in children with FXS, autism spectrum disorder (ASD), and typically developing controls. Parallel analysis in the FXS knockout mouse model will clarify its relevance to human FXS subgroups. In a randomized crossover study, we will evaluate the efficacy of closed-loop auditory entrainment, indexed on individual neurodynamic measures, aiming to normalize neural responses and enhance statistical learning performance. We anticipate this approach will yield opportunities to identify more effective early interventions that alter the trajectory of intellectual development in FXS.

Frontiers in NeuroscienceVol. 19
University of California, Riverside (US), Western University (CA), Cincinnati Children's Hospital Medical Center (US), Allen Institute for Brain Science (US), Brown University (US), Hospital Research Foundation (AU), University of Oklahoma Health Sciences Center (US), University of Cincinnati (US), University of Oklahoma (US), University of Cincinnati Medical Center (US)
National Institutes of Health
Openalex Percentile: Top 13%
Genetics and Neurodevelopmental Disorders
1.82
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