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Long-term neurocognitive and behavioral outcomes in patients with non-syndromic craniosynostosis
Jacqueline Ihnat1, Kevin Gao Hu1, Jeremy Goss1
1Division of Plastic Surgery, Department of Surgery, Yale School of Medicine, 330 Cedar Street, Boardman Building, New Haven, CT, 06510, USA.
Insights
Long-term cognitive function in craniosynostosis patients is comparable to the general population, but visual-motor integration deficits are noted. There is a higher prevalence of attention-deficit/hyperactivity disorder (ADHD) and autism behaviors.
Area of Science:
- Neuroscience
- Plastic Surgery
- Developmental Psychology
Background:
- Craniosynostosis is a birth defect involving the premature fusion of skull sutures.
- Early surgical correction is standard for non-syndromic craniosynostosis.
- Long-term outcomes regarding neurocognition and behavior require further investigation.
Purpose of the Study:
- To evaluate the long-term cognitive capabilities and behaviors of individuals who underwent corrective surgery for craniosynostosis in infancy.
- To identify potential neurodevelopmental challenges in late adolescence and early adulthood.
Main Methods:
- Retrospective identification of patients aged 16+ who had infant surgery for non-syndromic craniosynostosis.
- Standardized neurocognitive testing (BEERY, WASI) and behavioral assessments (CAARS-2, ASR, SRS-2, BRIEF) were administered.
- Data were analyzed to compare patient scores with general population norms.
Main Results:
- Neurocognitive scores (WASI FSIQ-4, PRI, VCI) were comparable to the general population.
- Significant deficits were observed in visual-motor integration (BEERY VMI, VP, MC).
- Higher prevalence of attention-deficit/hyperactivity disorder (ADHD) and autism spectrum disorder (ASD) traits were identified.
Conclusions:
- Individuals surgically treated for craniosynostosis exhibit typical general neurocognition but may have specific visual-motor integration deficits.
- There is an increased incidence of ADHD and autism-related behaviors in this cohort.
- Findings support targeted screening and early intervention for affected individuals and their families.
Purpose:
In this study, we explore the long-term cognitive capabilities and behaviors of patients in late adolescence and early adulthood who underwent corrective surgery for craniosynostosis as infants.
Methods:
Patients 16 years of age or older who had undergone surgery as infants for any type of non-syndromic craniosynostosis were identified from the operating records of three craniofacial plastic surgeons from a single institution. Participants underwent standardized neurocognitive testing using the BEERY and WASI. A subset of patients additionally underwent behavioral testing using the CAARS-2, ASR, SRS-2, and BRIEF standardized tests.
Results:
Thirty-two participants underwent neurocognitive testing and 22 participants underwent behavioral testing. The mean scores for the WASI FSIQ-4, PRI, and VCI were not significantly different than the general population. The mean score for BEERY VMI was 95, BEERY VP was 97, and BEERY MC was 92, all of which were significantly lower than the population average. Twenty-three percent of participants scored high or very high on the CAARS-2 ADHD index, and of the 44 total patients contacted, 13.6% either confirmed having a diagnosis of autism or tested moderate-severe on the SRS-2 autism screener.
Conclusions:
Patients have comparable neurocognition relative to the general population, with deficits in visual and motor integration. However, there is a significantly higher prevalence of ADHD and autism-related behaviors in this cohort. These findings are useful in counseling parents with infants who have been diagnosed with craniosynostosis, as well as in directing early screening and intervention for these patients to provide the resources and therapies families need.
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