Abstract

Children with single-suture craniosynostosis (SSC) have higher rates of learning disabilities and related neurocognitive problems than unaffected peers. Executive function (EF) and attention are thought to be areas of particular vulnerability, though studies to date have been limited by small sample sizes and a lack of control groups. We evaluated 179 school-aged children with SSC (cases) and 183 unaffected controls at an average age of 7 years using clinician-administered and parent and teacher report measures of EF and attention. Among children with SSC, we examined differences as a function of suture location (sagittal, metopic, unicoronal, or lambdoid) and age of corrective surgery. We used linear regression analyses, adjusted for potential confounders, to compare the two groups on all outcome measures. Cases scored lower than controls on most measures, though the magnitude of these differences was small and most were statistically insignificant. The largest relative deficit was on a measure of inhibitory control. Results changed little in sensitivity analyses adjusting for potential attrition bias and for the effects of developmental and academic interventions. Among cases, there were few differences in relation to the location of suture fusion or timing of surgery. Overall, we found limited evidence of broad deficits in EF or attention in children with SSC relative to unaffected controls. Neurocognitive development for children with SSC appears to be variable, across affected children and outcomes assessed. Further research is needed to understand the potential sources of this variability.

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