Predicting developmental delay in a longitudinal cohort of preschool children with single‐suture craniosynostosis: is neurobehavioral assessment important?

Abstract

AIM The aim of this study was to determine whether neurobehavioral assessment before and after cranial vault surgery can improve prediction of developmental delay in children with single-suture craniosynostosis (SSC), after accounting for clinical (SSC diagnosis and surgery age) and demographic ‘baseline’ variables. METHOD Children with SSC were referred by the treating surgeon or pediatrician before surgery. Neurobehavioral assessments were performed at ages of approximately 6, 18, and 36 months. Iterative models were developed to predict delay, as determined by one or more tests of cognitive, motor, and language skills at 36 months. We selected from groups of variables entered in order of timing (before or after corrective surgery), and source of information (parent questionnaire or psychometric testing). RESULTS Good predictive accuracy (as determined by area under the receiver operating characteristic curve [AUC]) was obtained with the baseline model (AUC=0.66), which incorporated age at surgery, sex, and socio-economic status. However, predictive accuracy was improved by including pre- and post-surgery neurobehavioral assessments. Models incorporating post-surgery neurobehavioral testing (AUC=0.79), pre-surgery testing (AUC=0.74), or both pre- and post-surgery testing (AUC=0.79) performed similarly. However, the specifity of all models was considered to be moderate (≤0.62). INTERPRETATION Prediction of delay was enhanced by assessment of neurobehavioral status. Findings provide tentative support for guidelines of care that call for routine testing of children with SSC.

Language, learning, and memory in children with and without single-suture craniosynostosis

“…Neurodevelopmental assessment at 18 months of age may help identify those children requiring closer follow-up through school age. 18 Finally, future research is needed to investigate language, memory, and learning for this population during the middle to high school years.…”

Parental Perceptions of Neurodevelopment in Toddlers Following Craniosynostosis Repair

“…When asked whether they agree that their child is different for the better other than appearance from 1 to 5 with 5 being ''strongly agree,'' the mean score was 3.8 (range [1][2][3][4][5].…”

“…1 Incidence rates vary by suture type with sagittal synostosis most common at 1 in 5000 births and lambdoid synostosis least common at 1 in 200,000 births. 2 Surgical repair performed before 12 months can be safe and provides a lasting aesthetic contour improvement.…”

Neurobehavioural assessment: a useful component of holistic care in single‐suture craniosynostosis

“…Gray et al. 's paper on the outcome of surgery for single‐suture craniosynostosis (SSC) for preschool children at the age of 3 years is an impressive example of collaboration between disciplines in clinical neuroscience that range from neurosurgery to behavioural paediatrics. It builds on a careful longitudinal study of children undergoing surgery for SSC enrolled across a number of US sites.…”