Related Experiment Video
Updated: May 25, 2026
Midface Hypoplasia and Cranial Base Morphology in Syndromic Craniosynostosis: A Comparative Analysis Study Using a Predictive Regression Model
Published on: November 4, 2025
Multicenter study of neurodevelopment in 3-year-old children with and without single-suture craniosynostosis
Jacqueline R Starr1, Brent R Collett, Rebecca Gaither
1Department of Epidemiology, University of Washington, Seattle, WA, USA.
Insights
Children with single-suture craniosynostosis show lower neurodevelopmental scores compared to peers. Early screening is recommended for these children to identify potential developmental delays.
Area of Science:
- Pediatric neurosurgery
- Developmental pediatrics
- Craniofacial anomalies
Background:
- Single-suture craniosynostosis is a condition where one of the sutures in a baby's skull fuses prematurely.
- Neurodevelopmental outcomes in affected children require further investigation.
Purpose of the Study:
- To compare neurodevelopmental scores in 3-year-old children with single-suture craniosynostosis versus children with normal suture development.
- To assess the hypothesis of lower neurodevelopmental outcomes in children with single-suture craniosynostosis.
Main Methods:
- A longitudinal comparison study was conducted across five tertiary care craniofacial centers.
- Children with single-suture craniosynostosis (cases) were frequency-matched with controls without craniosynostosis.
- Neurodevelopment was assessed using the Bayley Scales and Preschool Language Scales at baseline, 18, and 36 months.
Main Results:
- Children with single-suture craniosynostosis exhibited significantly lower mean neurodevelopmental scores (3-6 points deficit).
- The odds of developmental delay ranged from 1.5 to 2.0 compared to controls.
- Age at craniosynostosis repair did not strongly correlate with neurodevelopmental performance.
Conclusions:
- Children with single-suture craniosynostosis consistently demonstrate lower neurodevelopmental scores.
- These findings support the need for routine neurodevelopmental screening in this population.
- Early identification and intervention can potentially improve outcomes for affected children.
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.
