Radiographic severity associated with worse executive functioning in children with metopic craniosynostosis
Mariana Almeida*1, David Alper1, Carlos Barrero2, Mica C. Williams1, Jordan Swanson2, John Persing1, Michael Alperovich1
1Yale School of Medicine, New Haven, CT; 2Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA
Children with metopic synostosis are at risk for neurocognitive and behavioral deficits. The impact of radiographic severity has yet to be fully elucidated. We aimed to explore how radiographic severity, assessed manually and with artificial intelligence, influences both neurocognitive and behavioral outcomes at school age.
Children ages 6-18 years old with surgically corrected metopic synostosis underwent testing for intelligence quotient (IQ), academic achievement, and visuomotor integration. Parents completed behavioral assessments about their child: Conners-3 (ADHD), Social Responsiveness Scale-2 (autism spectrum disorder), Behavior Rating Inventory of Executive Function-2 (BRIEF-2: executive function), and Child's Behavioral Checklist (overall behavioral functioning). Endocranial bifrontal angle (EBA), adjusted EBA (aEBA), frontal angle (FA), and AI-derived metopic severity score (MSS) were determined. Multivariate linear regressions evaluated the association of age at surgery and severity with neurocognitive and behavioral outcomes.
There were 64 patients, with an average age at surgery of 9.4±7.6 months and average age at testing of 10.9±3.6 years. Many patients reached borderline clinical levels for symptoms of ADHD as assessed by the inattention (45.3%) and hyperactivity (42.2%) subscales of the Conners-3. Multivariate linear regression identified associations between the FA and verbal (p=0.01) and full-scale IQ (p=0.02). Age at surgery (p=0.02), MSS (p=0.02), EBA (p=0.02) and aEBA (p=0.02) were associated with poorer scores of the executive function subscale of the Conners-3. aEBA was associated with worse scores of executive functioning subscales, the cognitive regulation index (p=0.03) and global executive composite (p=0.05) of the BRIEF-2.
Many children with metopic synostosis have signs of ADHD. Pre-operative radiographic severity was associated with neurocognitive and behavioral outcomes at school age. A greater FA was associated with higher IQ scores. Later surgery and greater radiographic severity were associated with worse executive functioning.
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