Sex differences in resting EEG power in Fragile X Syndrome

Smith, Elizabeth G.; Pedapati, Ernest V.; Liu, Rui; Schmitt, Lauren; Dominick, Kelli C.; Shaffer, Rebecca C.; Sweeney, John A.; Erickson, Craig A.

doi:10.1016/j.jpsychires.2021.03.057

Cited by 30 publications

(48 citation statements)

References 41 publications

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“…Results of the replication study cohort also showed higher delta power and increased low gamma power. These results were expected as they confirm previous studies, supporting a robust signature of FXS resting-state EEG (20)(21)(22)27). Whereas, TBR was found to stabilize toward less discrepancies between theta and beta power with age, it did not differ between FXS and controls.…”

Section: Psd Et Tbrsupporting

confidence: 89%

“…An association between APF and language acquisition has been reported in the literature (66), suggesting that FXS participants who scored higher on the inappropriate speech subscale might have better language abilities in general, though their speech may be unsuitable for certain situations. Notably, alpha power and APF were not found altered in female FXS patients in previous studies (21,64). However, we did not find any sex differences in APF in our FXS sample.…”

Section: Psd Et Tbrcontrasting

confidence: 76%

“…Recent resting-state EEG studies have shown increased resting-state power in animal models of FXS, notably in delta and gamma frequency bands (18,19). Studies with FXS adults (20), male adults (21) and young boys (22) obtained similar results. Gamma frequency bands are associated with high-level cognitive functions in healthy controls while performing cognitively demanding tasks (20,23).…”

Section: Introductionmentioning

confidence: 65%

“…First, alpha peak frequency (APF), namely the frequency at which maximum power occurs within the alpha band, shifts from theta to alpha during brain maturation. Importantly, it was found to be altered in many neurodevelopmental disorders, including attention deficit hyperactivity disorder (ADHD), ASD, and FXS ( 21 , 28 , 30 ).…”

Section: Introductionmentioning

confidence: 99%

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EEG Signal Complexity Is Reduced During Resting-State in Fragile X Syndrome

et al. 2021

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Introduction: Fragile X syndrome (FXS) is a genetic disorder caused by a mutation of the fragile X mental retardation 1 gene (FMR1). FXS is associated with neurophysiological abnormalities, including cortical hyperexcitability. Alterations in electroencephalogram (EEG) resting-state power spectral density (PSD) are well-defined in FXS and were found to be linked to neurodevelopmental delays. Whether non-linear dynamics of the brain signal are also altered remains to be studied.Methods: In this study, resting-state EEG power, including alpha peak frequency (APF) and theta/beta ratio (TBR), as well as signal complexity using multi-scale entropy (MSE) were compared between 26 FXS participants (ages 5–28 years), and 7 neurotypical (NT) controls with a similar age distribution. Subsequently a replication study was carried out, comparing our cohort to 19 FXS participants independently recorded at a different site.Results: PSD results confirmed the increased gamma, decreased alpha power and APF in FXS participants compared to NT controls. No alterations in TBR were found. Importantly, results revealed reduced signal complexity in FXS participants, specifically in higher scales, suggesting that altered signal complexity is sensitive to brain alterations in this population. The replication study mostly confirmed these results and suggested critical points of stagnation in the neurodevelopmental curve of FXS.Conclusion: Signal complexity is a powerful feature that can be added to the electrophysiological biomarkers of brain maturation in FXS.

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Section: Psd Et Tbrsupporting

confidence: 89%

Section: Psd Et Tbrcontrasting

confidence: 76%

Section: Introductionmentioning

confidence: 65%

Section: Introductionmentioning

confidence: 99%

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EEG Signal Complexity Is Reduced During Resting-State in Fragile X Syndrome

et al. 2021

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“…This is of critical importance given that while FXS is a single gene disorder, the clinical presentations of the disorder vary widely. Additionally, we have demonstrated that the EEG signatures in FXS differ based on sex (44), and therefore it is possible that EEG target engagement also may differ based upon the sex of the subject. Thus, increasing our human sample size in this line of research will be essential to answer these questions of importance to the eld.…”

Section: Discussionmentioning

confidence: 98%

Baclofen-Associated Neurophysiologic Target Engagement Across Species in Fragile X Syndrome

Jonak¹,

Pedapati²,

Schmitt³

et al. 2021

Preprint

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Background: Fragile X Syndrome (FXS) is the most common inherited form of neurodevelopmental disability. It is often characterized, especially in males, by intellectual disability, anxiety, repetitive behavior, social communication deficits, delayed language development and abnormal sensory processing. Recently, we identified electroencephalographic (EEG) biomarkers that are conserved between the mouse model of FXS (Fmr1 KO mice) and humans with FXS. Methods: In this report, we evaluate small molecule target engagement utilizing multielectrode array electrophysiology in the Fmr1 KO mouse and in humans with FXS. Neurophysiologic target engagement was evaluated using single doses of the GABAB selective agonist racemic baclofen (RBAC). Results: In Fmr1 KO mice and in humans with FXS, baclofen use was synchronously associated with suppression of elevated gamma power and increase in theta power at rest. In the Frm1 KO mice, a baclofen-associated improvement in auditory chirp synchronization was also noted. Conclusions: Overall, we noted synchronized target engagement of RBAC on resting state electrophysiology, in particular the reduction of aberrant high frequency gamma activity, across species in FXS. This finding holds promise for translational medicine approaches to drug development for FXS, synchronizing treatment study across species using well-established EEG biological markers in this field. Trial Registration: The human experiments are registered under NCT02998151.

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