Date of Award

January 2024

Document Type

Open Access Thesis

Degree Name

Medical Doctor (MD)

Department

Medicine

First Advisor

Michael Alperovich

Abstract

Children with metopic nonsyndromic craniosynostosis have been found to have more neurocognitive and behavioral difficulties, when compared to other types of nonsyndromic craniosynostosis. These neurocognitive and behavioral difficulties can be apparent later in life even following surgical correction of the cranial dysmorphology at infancy. The factors that may be contributing to these difficulties have not been fully elucidated. In metopic craniosynostosis, radiographic severity can be measured using computed tomography imaging. Pre-operative radiographic severity has been demonstrated to be associated with worse reading abilities at school age. However, these studies have had limited sample sizes. Furthermore, the impact of radiographic severity on behavior has not been explored. We aimed to investigate the impact of severity on neurocognitive and behavioral outcomes at school age. Children 6-18 years old with surgically corrected metopic nonsyndromic craniosynostosis underwent behavioral assessment and neurocognitive testing. Parents completed four behavior assessments about their child, which included the Conners-3 (ADHD), Social Responsiveness Scale-2 (autism spectrum disorder), Behavior rating Inventory of Executive Function-2 (BRIEF-2: executive function) and Child’s Behavior Checklist (overall behavior). Neurocognitive testing was completed virtually which included assessment of intelligence quotient, academic achievement and visuomotor integration. Radiographic measurements were obtained from pre-operative computed topography images. The measurements included the endocranial bifrontal angle (EBA), adjusted EBA (aEBA), frontal angle (FA) and AI-derived metopic severity score (MSS). Multivariable linear regressions were used to evaluate the association between pre-operative radiographic severity with behavioral and neurocognitive outcomes while controlling for sociodemographic factors. There were 87 children with pre-operative radiographic images and neurocognitive testing (average age 10.9 ± 3.3 years.), with 67 children who also had completed behavioral assessments. Multivariable regression demonstrated that a smaller FA, aEBA and EBA, which indicate greater severity was associated with worse scores on all the subscales of the BRIEF-2, which evaluates executive functioning, and the executive subscale of the Conners-3 (p<0.05). Furthermore, all measurements of severity were also associated with worse scores on the rule-breaking subscale of the CBCL. Lastly, multivariable analysis demonstrated a significant association between the FA and worse perceptual IQ scores of the WASI-II (B=1.16, p<0.05). Children with greater pre-operative severity of metopic synostosis tend to have worse executive function at school age, even after surgical intervention and correction. There is less of an association between radiographic severity and neurocognitive assessments, given that only one of the regression models demonstrated significant associations. Most children at school age have signs and symptoms of ADHD that would warrant further assessment for diagnosis. Future research should aim at identifying the direct structural changes to the brain and how this may impact behavioral outcomes in children.

Comments

This is an Open Access Thesis.

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