Long-Term Behavioral Outcomes are Different Between School-Aged Children with Metopic and Sagittal Synostosis
David P. Alper*, Mariana Almeida, Mica C. Williams, Vikram Mookerjee, Heloise M. de Baun, John Persing, Michael Alperovich
Division of Plastic Surgery, Department of Surgery, Yale School of Medicine, New Haven, CT
Nonsyndromic craniosynostosis has been associated with neurocognitive and behavioral deficits. However, small sample sizes, variability in age, and assessment metrics have limited conclusions. We aimed to compare behavioral impairments of school-aged children with metopic synostosis (MS) and sagittal synostosis (SS) using a battery of behavioral assessments.
Parents of children ages 6-18 years old with MS and SS completed the Child’s Behavior Checklist (CBCL: overall behavioral functioning), Social Responsiveness Scale-2 (SRS-2: autism spectrum disorder [ASD]), Conners-3 (ADHD), and the Behavior Rating Inventory of Executive Function-2 (BRIEF-2: executive function). ANCOVA was used to assess for differences while controlling for sociodemographic risk, age at surgery, surgery type and IQ. Logistic regression was used to determine factors associated with reaching borderline clinical levels.
106 parents completed the assessments (60 MS, 46 SS). Children with MS were older at the time of assessment (10.41±3.60 vs 8.43±2.13 years, p<0.001). There was no difference in age at surgery (MS: 9.33±7.79 vs SS: 6.83±5.07 months, p=0.06). More children with MS had characteristics of ASD, demonstrated by reaching borderline clinical levels of the restricted interests and repetitive behavior (SRS-2: 30.0% vs 10.9%, p=0.01) subscale. More children with MS had worse executive function, assessed by the emotional regulation index (33.3% vs 17.4%, p=0.03) and global executive composite (31.7% vs 17.4%, p=0.05). SS had greater problems with somatic (57.59±0.99 vs 54.89±0.91, p=0.05), rule breaking (55.33±0.80 vs 52.53±0.74, p=0.02) and externalizing problem (52.32±1.90 vs 46.88±1.75, p=0.04). Greater age at surgery was associated with borderline clinical scores of ASD characteristics, such as social cognition (p=0.05) and social motivation (p=0.04) and emotional regulation (p=0.01).
Children with MS and SS were found to have varying behavioral difficulties. Children with MS had more problems in executive function, while children with SS had more problems with externalizing behaviors.
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