Abstract
BACKGROUND: Single-suture craniosynostosis (SSC) results in head shape anomalies that likely affect social perceptions of appearance. The purpose of this study was to evaluate laypersons' ratings of attractiveness in children with and without SSC. Among cases, we also examined differences by suture fused and age at surgery. METHODS: We collected photographs of 196 children with SSC and 186 children without SSC as infants (before surgery, for cases) and at ages 18 and 36 months. Photographs were rated by 8 raters, who were blinded to the population being studied. We used linear regression to compare appearance ratings for the 2 groups at each visit and to evaluate changes over time. Regression analyses were used to examine the association between age at surgery and appearance ratings. RESULTS: Children with SSC received lower appearance ratings than unaffected controls at each visit (all P < 0.001). Appearance ratings decreased over time, with a similar trajectory for children with and without SSC. Among cases, those with unicoronal and lambdoid synostosis had the lowest ratings and those with sagittal synostosis had the highest. Age at surgery was inversely associated with appearance ratings. CONCLUSIONS: Children with SSC received lower appearance ratings than unaffected controls, with minimal change after surgery. Better outcomes were associated with earlier surgery. These findings do not indicate that children with SSC failed to benefit from surgery, as without surgical intervention, asymmetrical head shape would likely have worsened over time. However, our data suggest that appearance does not fully "normalize."