Early Longitudinal Development in a Diverse Prospective Cohort with Sagittal,Unicoronal,or Metopic Craniosynostosis

Abstract

Objective

To describe early development in children with nonsyndromic craniosynostosis (CS) compared to norms, longitudinally, and by surgery age.

Design

Prospective longitudinal cohort study.

Participants

Patients with CS who had open approach single-stage surgery with the same craniofacial surgeon and neurosurgeon 2009-2019.

Main Outcome Measure

Bayley Scales of Infant and Toddler Development—Third Edition (Bayley-III) at Time 1 (preoperative), Time 2 (6-months postoperative), Time 3 (18-months postoperative), and Time 4 (age 36 months).

Results

Participants (N = 268) had sagittal (50%), unicoronal (26%), or metopic (24%) CS and were mostly Latino (49%) males (67%) with public insurance (57%). The mean surgical age was 7.9 ± 4.3 months with 91% of surgeries completed by 12 months. Higher scores were associated with female sex, higher socioeconomic status, private insurance, and US-born fathers. All mean Bayley-III scores were in the average range clinically and most scores were similar to test norms. Average rates of participants with a delay (<10th percentile) across time points included: Metopic (27%), sagittal (20%), and unicoronal (15%). Development was generally stable over time. There was no relationship between surgery age and development within the context of only 9% of surgeries completed past 12 months.

Conclusions

Sociodemographic influences on development can help identify possible protective and risk factors. While mean performance was consistently in the average clinical range, delays were seen in nearly a third of metopic CS, a fifth of sagittal CS, and 15% of unicoronal CS. Ongoing screening can facilitate appropriate developmental supports, most often for language and gross motor skills.