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Early Longitudinal Development in a Diverse Prospective Cohort with Sagittal, Unicoronal, or Metopic Craniosynostosis
Alexis L Johns1,2, J Gordon McComb3,4, Mark M Urata1,5
1Division of Plastic and Maxillofacial Surgery, Children's Hospital Los Angeles, Los Angeles, CA, USA.
Insights
Children with craniosynostosis (CS) show average early development, but delays are common, especially in metopic CS. Ongoing screening is crucial for developmental support in language and motor skills.
Area of Science:
- Pediatric Surgery
- Developmental Pediatrics
- Craniofacial Anomalies
Background:
- Nonsyndromic craniosynostosis (CS) involves premature fusion of skull sutures, potentially impacting neurodevelopment.
- Early surgical intervention is standard, but longitudinal developmental outcomes require further elucidation.
Purpose of the Study:
- To longitudinally assess early development in children with nonsyndromic CS.
- To compare development to normative data and analyze outcomes based on surgical timing and sociodemographic factors.
Main Methods:
- Prospective longitudinal cohort study of 268 children with CS undergoing open-approach, single-stage surgery.
- Development assessed using the Bayley Scales of Infant and Toddler Development-Third Edition (Bayley-III) preoperatively and at 6, 18, and 36 months postoperatively.
Main Results:
- Mean Bayley-III scores were consistently in the average clinical range and similar to test norms.
- Developmental delays (<10th percentile) were observed in 27% of metopic, 20% of sagittal, and 15% of unicoronal CS cases.
- Higher scores correlated with female sex, higher socioeconomic status, private insurance, and US-born fathers.
Conclusions:
- While mean development is average, significant proportions of children with CS experience delays, particularly in language and gross motor skills.
- Sociodemographic factors influence development, highlighting the need for targeted screening and support.
- Developmental trajectories were generally stable, with no clear link to early surgical timing given the cohort's early intervention pattern.
Abstract:
ObjectiveTo describe early development in children with nonsyndromic craniosynostosis (CS) compared to norms, longitudinally, and by surgery age.DesignProspective longitudinal cohort study.ParticipantsPatients with CS who had open approach single-stage surgery with the same craniofacial surgeon and neurosurgeon 2009-2019.Main Outcome MeasureBayley 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).ResultsParticipants (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.ConclusionsSociodemographic 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.
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