Neurologic Characterization of Craniosynostosis: Can Direct Brain Recordings Predict Language Development?
Robin T. Wu, BS, Paul F. Abraham, BS, James Nie, BS, Taylor Halligan, BS, Kyle S. Gabrick, MD, Rajendra F. Sawh-Martinez, MD MHS, Derek M. Steinbacher, MD DMD, Michael A. Alperovich, MD MSc, John A. Persing, MD.
Yale University School of Medicine, New Haven, CT, USA.
BACKGROUND: Early detection and prevention is essential for language remediation in non-syndromic craniosynostosis (NSC). The current standard assessment, the Bayley Scales of Infant Development (BSID), provides little predictive value for long-term development. Auditory event-related potentials (ERPs), in particular the mismatch negativity (MMN), measure passive neurological responses to speech sounds, suggesting a promising avenue for analyzing speech development. We provide long-term validation of ERP testing and follow-up analysis of differences between subtypes of NSC.
METHODS: NSC infants were recruited pre and post-operatively. Participants were given the BSID by a child psychologist and then were presented with a non-native phoneme discrimination paradigm. Auditory stimuli were set at 80 dB, and EEG was recorded at 250 Hz with a 128-channel HydroCel Geodesic Sensor Net. The MMN component was the largest negative amplitude in the difference wave between 80-300ms after stimulus presentation. Once patients reached >6 years of age, they completed a battery of neurodevelopmental tests (Wechsler Abbreviated Scale of Intelligence and Wechsler Fundamentals). Statistical analysis involved correlations and t-tests.
RESULTS: 10 NSC patients with ERP testing in infancy were neurocognitively tested (average age 7.9 years; 30% female; 50% SSO, 20% MSO, 20% ULC). Correlation analyses showed left frontal cluster MMN strongly correlated with word-reading (R=0.713, p=0.031), reading-comprehension (R=0.745, p=0.021), and language-composite scores (R=0.771, p=0.015). BSID scores had no predictive value (R<0.5, p>0.05). 18 SSO, 17 MSO, 6 CSO, and 39 normal control patients were included in the infant ERP analysis. Pre-operatively, SSO and MSO patients had attenuated left frontal MMN amplitudes compared to controls (p=0.003). Post-operatively, attenuated responses were no longer presented in the SSO cohort while MSO patients retained attenuations (p=0.041). CSO patients were comparable to controls pre and post-operatively. With inter-group comparison, MSO patients had post-operative attenuations in left frontal MMN amplitudes when compared to SSO (p=0.041).
CONCLUSIONS: Our prospective longitudinal assessment showed that ERP assessment in NSC patients has significantly better predictive value for future neurocognitive assessment than the current gold standard BSID test. Left frontal measurements approximate the location of language-associated brain centers. Pre-operatively, all three groups had similar language profiles. However, following significant improvements in the SSO group and no changes in the MSO group, SSO patients had improved left brain language acquisition in comparison to MSO. This may help tailor treatment for possible language deficits in future development.
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