Multicenter study of neurodevelopment in 3-year-old children with and without single-suture craniosynostosis

Abstract

Objective: To evaluate the hypothesis that 3-year-old children with single-suture craniosynostosis would receive lower neurodevelopmental scores than a comparable group of children born with patent sutures.

Design: Longitudinal comparison study.

Setting: Five tertiary care craniofacial centers.

Participants: Patients with craniosynostosis (cases) and a comparison group of children without craniosynostosis(controls). Patients diagnosed with single-suture craniosynostosis from 2002 to 2006 were eligible as cases.Controls were frequency-matched to cases on age, sex, race, socioeconomic status, and study site.

Main exposure: Craniosynostosis.

Main outcome measures: We administered the Bayley Scales of Infant Development, Second Edition, mental and motor development indices and the Preschool Language Scales, Third Edition, receptive and expressive communication scales. Children were evaluated at baseline (before surgery in cases and at a similar age in controls)and at 18 and 36 months of age. We compared the groups' performances at 36 months by fitting adjusted linear and logistic regression models. We also estimated adjusted associations between age at surgery and neurodevelopmental scores.

Results: Adjusted mean case deficits ranged from 3 to 6 points (P≤ .008 for all comparisons). Compared with controls, the odds of cases being delayed ranged from 1.5 to 2.0, depending on the neurodevelopmental scale (P values ranged from .03 to .09). Cases' ages at craniosynostosis repair were not strongly related to neurodevelopmental performance.

Conclusions: In this large, carefully controlled, multicenter study, we observed consistently lower mean neurodevelopmental scores in children with single-suture craniosynostosis compared with controls. These results provide further support for neurodevelopmental screening in young children with single-suture craniosynostosis.