Development of Neural Response to Novel Sounds in Fragile X Syndrome: Potential Biomarkers

Auditory processing abnormalities in fragile X syndrome (FXS) may contribute to difficulties with language development, pattern identification, and contextual updating. Participants with FXS (N = 41) and controls (N = 27) underwent auditory event-related potentials during presentation of an oddball...

Full description

Saved in:
Bibliographic Details
Published in:American journal on intellectual and developmental disabilities 2020-11, Vol.125 (6), p.449-464
Main Authors: Ethridge, Lauren, Thaliath, Andrew, Kraff, Jeremy, Nijhawan, Karan, Berry-Kravis, Elizabeth
Format: Article
Language:English
Subjects:
Age
Auditory Perception
Auditory Stimuli
Behavior Rating Scales
Biomarkers
Caregivers
Clinical trials
Cognitive Processes
Electroencephalography
Executive function
Gender
Gender Differences
Genetic Disorders
Habituation
Intellectual disabilities
Language
Language Acquisition
Language Skills
Neurological Impairments
Novelty (Stimulus Dimension)
Sensors
Sensory perception
Citations: Items that this one cites
Items that cite this one
Online Access:Get full text
Tags: Add Tag
No Tags, Be the first to tag this record!
Description
Summary:Auditory processing abnormalities in fragile X syndrome (FXS) may contribute to difficulties with language development, pattern identification, and contextual updating. Participants with FXS (N = 41) and controls (N = 27) underwent auditory event-related potentials during presentation of an oddball paradigm. Data was adequate for analysis for 33 participants with FXS and 27 controls (age 4-51 y, 13 females [FXS]; 4-54 y, 11 females [control]). Participants with FXS showed larger N1 and P2 amplitudes, abnormal lack of modulation of P1 and P2 amplitudes and P2 latency in response to oddball stimuli ) relative to controls: Females with FXS were more similar to controls. Participants with FXS showed a marginal speeding of the P2 latency, suggesting potentiation to oddball stimuli rather than habituation. Participants with FXS showed a heightened N1 habituation effect compared to controls. Gamma power was significantly higher for participants with FXS. Groups did not differ on mismatch negativity. Both controls and participants with FXS showed similar developmental trajectories in P1 and N1 amplitude, P2 latency, and gamma power, but not for P2 amplitude. One month retest analyses performed in 14 participants suggest strong test-retest reliability for most measures. Individuals with FXS show previously demonstrated increased response amplitude and high frequency neural activity. Despite an overall normal developmental trajectory for most measures, individuals with FXS show age-independent but gender-dependent decreases in complex processing of novel stimuli. Many markers show strong retest reliability even in children and thus are potential biomarkers for clinical trials in FXS.
ISSN:1944-7515
1944-7558
DOI:10.1352/1944-7558-125.6.449