ABSTRACT: To evaluate the hypothesis that 3-year-old children with single-suture craniosynostosis would receive lower neurodevelopmental scores than a comparable group of children born with patent sutures.
Longitudinal comparison study.
Five tertiary care craniofacial centers.Participants: Patients with craniosynostosis (cases) and a comparison group of children without craniosynostosis(controls). Patients diagnosed with single-suture craniosynostosis from 2002 to 2006 were eligible as cases.Controls were frequency-matched to cases on age, sex, race, socioeconomic status, and study site.
We administered the Bayley Scales of Infant Development, Second Edition, mental and motor development indices and the Preschool Language Scales, Third Edition, receptive and expressive communication scales. Children were evaluated at baseline (before surgery in cases and at a similar age in controls)and at 18 and 36 months of age. We compared the groups' performances at 36 months by fitting adjusted linear and logistic regression models. We also estimated adjusted associations between age at surgery and neurodevelopmental scores.
Adjusted mean case deficits ranged from 3 to 6 points (P≤ .008 for all comparisons). Compared with controls, the odds of cases being delayed ranged from 1.5 to 2.0, depending on the neurodevelopmental scale (P values ranged from .03 to .09). Cases' ages at craniosynostosis repair were not strongly related to neurodevelopmental performance.
In this large, carefully controlled, multicenter study, we observed consistently lower mean neurodevelopmental scores in children with single-suture craniosynostosis compared with controls. These results provide further support for neurodevelopmental screening in young children with single-suture craniosynostosis.
Archives of pediatrics & adolescent medicine 02/2012; 166(6):536-42. · 3.73 Impact Factor
ABSTRACT: Objective : To compare relative levels of stress reported by mothers and fathers in families containing infants with and without single-suture craniosynostosis. Design : Case-control study. Participants : Mothers and fathers of 246 infants with recently diagnosed single-suture craniosynostosis and 253 frequency-matched control infants completed the Parenting Stress Index just prior to their infant's cranioplastic surgery. Family demographic information and mothers' ratings of the severity of their child's single-suture craniosynostosis were obtained. Results : Average Parent Domain scores for parents of infants with single-suture craniosynostosis differed little from those reported by parents of control infants; however, Child Domain scores among parents of infants with single-suture craniosynostosis were higher on some subscales, primarily related to unexpected infant health and appearance issues. In both groups, fathers reported higher Child Domain stress than mothers, and mothers reported higher Parent Domain stress than fathers. Case mothers reported greater stress if they perceived their child's condition as more noticeable to others. Conclusions : Prior to cases' cranioplastic surgery, parents of children with and without single-suture craniosynostosis reported similar levels of stress in relation to their parenting roles and the behavioral characteristics of their infants. Visibility of condition should be considered a risk for increased stress for mothers of infants with single-suture craniosynostosis. Stress differences between mothers and fathers were far more discernible than those associated with the presence or absence of single-suture craniosynostosis.
The Cleft Palate-Craniofacial Journal 09/2011; 48(5):509-18. · 0.82 Impact Factor
ABSTRACT: Although most infants with single-suture craniosynostosis (SSC) appear to have neurodevelopmental test scores in the average range, SSC has been associated with cognitive and motor delays during infancy. Whether and when surgery improves such deficits are not yet known. The authors aimed to compare the pre- and postsurgical neurodevelopmental status of patients with SSC with those of control infants without craniosynostosis.
The authors conducted a large, multicenter, longitudinal study of 168 infants with craniosynostosis and 115 controls without synostosis who were of similar age, race, sex, and socioeconomic status. The authors assessed participants by using the Bayley Scales of Infant Development, Second Edition (BSID-II) and the Preschool Language Scale, Third Edition (PLS-3) at enrollment, before patients' intracranial surgery, and when participants were 18 months of age (after surgery for patients).
After adjusting for potential confounding factors in linear regression analyses, the authors found a tendency for patients to perform similarly to or slightly worse than controls on neurodevelopmental examinations at both visits. After surgery, the patients' mean scores were 0.6 to three points lower than those of controls on the five BSID-II and PLS-3 scales (p = 0.02-0.07). Compared with controls, patients had 2.3 and 1.9 times the adjusted odds of scoring in the delayed range on either BSID-II scale (Mental Development Index and Psychomotor Development Index) for the first and second visits, respectively (p = 0.001 and p = 0.015, respectively). The patients' mean adjusted test scores were nearly unrelated to the timing of their surgery.
These findings support recommendations for neurodevelopmental screening in infants with SSC. Longer follow-up, as is being conducted with the patients in the present study, will be critical for identifying the potential longer-term correlates of SSC and its surgical correction.
Journal of Neurosurgery 09/2007; 107(2 Suppl):103-10. · 2.96 Impact Factor