This project will investigate the association between neurodevelopmental impairment and single suture craniosynostosis (SSC), defined as the premature fusion of a single cranial suture. This cohort of participants with SSC (n=240) and controls (n=240) were recruited in infancy (R01 DE 013813) and assessed at three time points: (1) prior to cranioplasty (age 6 months on average);(2) age 18 months;and (3) age 3 years. The long term objective of this project is to better understand how the developing cranium affects human growth and function. Existing studies of school-age children with SSC (primarily case record reviews or evaluations of small clinical samples) suggest a substantially elevated risk of learning, attention and behavior problems. Clarification of the impact of SSC is an important public health objective because families currently receive conflicting information regarding the rationale for cranioplasty. Some health providers believe that this procedure is only necessary to reduce craniofacial disfigurement, while others believe that the procedure prevents or minimizes neurodevelopmental disorder and brain deformation.
Specific aims are to: 1) Compare the neurodevelopment of school-age children with and without SSC across seven domains of neuropsychological functioning (intelligence, language abilities, attention and executive function, learning and memory, visual-motor skills, processing speed, and academic achievement);2) Develop predictive models of functional outcomes for children with SSC at age 7 using data obtained during infancy/toddler assessments;3) Examine neurobehavioral development as a function of pre- to post-surgery change in severity of synostosis, as determined by quantification of CT scans;4) Test the hypothesis that children with SSC will show greater degree of structural abnormality and decreased brain volumes on MRI when compared to age-matched controls.

Agency
National Institute of Health (NIH)
Institute
National Institute of Dental & Craniofacial Research (NIDCR)
Type
Research Project (R01)
Project #
5R01DE013813-10
Application #
8294403
Study Section
Special Emphasis Panel (ZDE1-SK (61))
Program Officer
Riddle, Melissa
Project Start
2000-07-01
Project End
2014-06-30
Budget Start
2012-07-01
Budget End
2014-06-30
Support Year
10
Fiscal Year
2012
Total Cost
$588,244
Indirect Cost
$231,303
Name
Seattle Children's Hospital
Department
Type
DUNS #
048682157
City
Seattle
State
WA
Country
United States
Zip Code
98105
Kapp-Simon, Kathleen A; Wallace, Erin; Collett, Brent R et al. (2016) Language, learning, and memory in children with and without single-suture craniosynostosis. J Neurosurg Pediatr 17:578-88
Birgfeld, Craig B; Dufton, Lynette; Naumann, Heather et al. (2015) Safety of Open Cranial Vault Surgery for Single-Suture Craniosynostosis: A Case for the Multidisciplinary Team. J Craniofac Surg 26:2052-8
Gray, Kristen E; Cradock, Mary Michaeleen; Kapp-Simon, Kathleen A et al. (2015) Longitudinal analysis of parenting stress in mothers and fathers of infants with and without single-suture craniosynostosis. Cleft Palate Craniofac J 52:3-11
Gray, Kristen E; Kapp-Simon, Kathleen A; Starr, Jacqueline R et al. (2015) Predicting developmental delay in a longitudinal cohort of preschool children with single-suture craniosynostosis: is neurobehavioral assessment important? Dev Med Child Neurol 57:456-62
Speltz, Matthew L; Collett, Brent R; Wallace, Erin R et al. (2015) Intellectual and academic functioning of school-age children with single-suture craniosynostosis. Pediatrics 135:e615-23
Cradock, Mary Michaeleen; Gray, Kristen E; Kapp-Simon, Kathleen A et al. (2015) Sex differences in the neurodevelopment of school-age children with and without single-suture craniosynostosis. Childs Nerv Syst 31:1103-11
Lam, Irma; Cunningham, Michael; Birgfeld, Craig et al. (2014) Quantification of skull deformity for craniofacial research. Conf Proc IEEE Eng Med Biol Soc 2014:758-61
Lam, Irma; Cunningham, Michael; Speltz, Matthew et al. (2014) Classifying Craniosynostosis with a 3D Projection-Based Feature Extraction System. Proc IEEE Int Symp Comput Based Med Syst 2014:215-220
Collett, Brent R; Gray, Kristen E; Kapp-Simon, Kathleen A et al. (2013) Laypersons' ratings of appearance in children with and without single-suture craniosynostosis. J Craniofac Surg 24:1331-5
Cloonan, Yona K; Collett, Brent; Speltz, Matthew L et al. (2013) Psychosocial outcomes in children with and without non-syndromic craniosynostosis: findings from two studies. Cleft Palate Craniofac J 50:406-13

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