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Midface Hypoplasia and Cranial Base Morphology in Syndromic Craniosynostosis: A Comparative Analysis Study Using a Predictive Regression Model
Published on: November 4, 2025
Neurodevelopmental and Psychiatric Outcomes in Pediatric Nonsyndromic Craniosynostosis: Insights for Plastic Surgery
Viraj N Govani1, Mary C Wilding, Janina Kueper
1Department of Plastic Surgery, University of Pittsburgh School of Medicine, Pittsburgh, PA.
Insights
Early diagnosis of craniosynostosis is crucial for pediatric neurodevelopmental outcomes. Delayed diagnosis correlates with a higher incidence of neurodevelopmental disorders, including autism spectrum disorder (ASD) and ADHD.
Area of Science:
- Pediatric Neurosurgery
- Neurodevelopmental Pediatrics
- Medical Genetics
Background:
- Long-term neurodevelopmental outcomes in craniosynostosis patients remain uncertain despite surgical advances.
- This study investigates the link between craniosynostosis clinical features and neurodevelopmental comorbidities.
Purpose of the Study:
- To examine the association between clinical presentation of craniosynostosis and neurodevelopmental outcomes.
- To identify factors influencing neurodevelopmental comorbidities in pediatric craniosynostosis patients.
Main Methods:
- Retrospective review of 655 pediatric patients with nonsyndromic craniosynostosis (2003-2023).
- Analysis included demographics, clinical presentation, neurocognitive diagnoses, surgical history, and outcomes.
- Statistical analyses (descriptive statistics, chi-square, t-tests, logistic regression) were performed.
Main Results:
- 121 patients (18.5%) had a neurodevelopmental diagnosis (ASD, ADHD, learning disorder, developmental delay), with 74 having multiple diagnoses.
- Delayed craniosynostosis diagnosis was significantly associated with increased odds of neurodevelopmental diagnoses (2% increase per month delayed, P < 0.001).
- Older age at diagnosis and first PRS visit correlated with higher rates of neurodevelopmental diagnoses; age at surgery was not significant.
Conclusions:
- Early diagnosis of craniosynostosis is critical, as delays are linked to a higher frequency of neurodevelopmental disorders in pediatric patients.
- The study highlights the importance of timely detection for improving neurodevelopmental outcomes.
- Further research is needed to clarify the causal relationship, particularly with systematic syndrome classification.
Introduction:
Despite advances in surgical treatment, uncertainty remains regarding the long-term neurodevelopmental outcomes of patients with craniosynostosis. This study aimed to investigate associations between clinical features of craniosynostosis and multiple neurodevelopmental comorbidities.
Methods:
We retrospectively reviewed patients with nonsyndromic craniosynostosis at a single institution (2003-2023), examining demographics, clinical presentation, neurocognitive diagnoses, surgeries, and patient outcomes. Descriptive statistics, chi-square tests, t tests, and logistic regressions were performed using STATA ( P < 0.05).
Results:
We analyzed 655 pediatric craniosynostosis patients with a median follow-up of 50 (21-90) months. Overall, 64.3% were male, 83.0% were White, and the median age at diagnosis of craniosynostosis was 11.03 (3.65-43.89) months. A total of 121 patients had a neurodevelopmental diagnosis (ASD, ADHD, learning disorder, or developmental delay), including 74 with multiple diagnoses.Neurodevelopmental diagnoses were associated with later craniosynostosis diagnosis (57.62 ± 44.56 vs 30.10 ± 41.21; P < 0.001), older age at first PRS visit (48.63 ± 40.75 vs 29.15 ± 41.21; P < 0.001), and longer PRS follow-up (81.70 ± 54.71 vs 57.73 ± 47.58; P = 0.002). In models adjusted for age at initial visit, surgical intervention, insurance type, race, sex, and suture location, each additional month of age at CS diagnosis was independently associated with a 2% increase in the odds of any neurodevelopmental outcome ( P < 0.001). Age at surgery was not significantly associated.
Conclusion:
This study underscores the critical importance of early diagnosis in craniosynostosis, as delayed detection was associated with increased frequency of a composite outcome of 4 common neurodevelopmental disorders in pediatric patients. However, without systematic syndrome classification, the causal nature of this relationship remains uncertain and warrants further investigation.
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