THE NORTH CAROLINA CLEFT OUTCOMES STUDY

DESCRIPTION (provided by applicant): Project Summary We propose a four-year study examining patterns of short and long-term health outcomes, health care use, and costs among children with and without orofacial clefts (OFC). The overarching goal of this project is to identify new opportunities for intervention to improve educational, psychological/behavioral, and surgical outcomes for children with OFC. The primary objective is to describe patterns and factors associated with short and long-term health outcomes of children with OFC, with a special focus on academic achievement (reading, writing, and math), behavioral/psychological outcomes (attention-deficit hyperactivity disorder (ADHD), family impact), and referral to and use of genetic and early intervention services. These outcomes will be compared to children without OFC. A secondary objective is to assess timeliness of services, including speech and language and genetic services, surgical outcomes, use and costs of team care, out of pocket expenses, and caregiver costs among families of children with OFC. This study will involve a longitudinal, retrospective and prospective case control study of North Carolina resident children born 2001 through 2013. The potential sample size will be about 4000 children ranging from 0 to 14 years old. Approximately 2000 children with non- syndromic OFC (cases) will be identified through the state's birth defects registry. Approximately 2000 children without birth defects (controls) will be matched by birth cohort. We will examine individual (maternal/caregiver and child) and system factors associated with the development of each of these outcomes and conditions. If applicable, we will match on certain characteristics, such as age, gender, and school grade. We will conduct these evaluations by linking several administrative data sources: the North Carolina birth defects registry, early intervention data, state-wide education data, vital statistics, health services, Medicaid enrollment records and paid claims, and clinically relevant information. We will conduct a comprehensive assessment of families' experiences at two different time points to better assess short and long-term outcomes. The survey will include questions about academic achievement, maternal bonding, family impact, developmental disabilities, experiences with team care, surgical procedures, satisfaction with surgery, and caregiver and out-of-pocket costs. Analytical approaches will include multivariate analyses to identify: prevalence and predictors of behavioral/psychological outcomes, academic achievement, timeliness and use of services (genetics services, speech and language, lip/palate repair and revision surgeries), expenditures, and use and costs of team care. Leading experts in the field from the two primary craniofacial and cleft centers in the state, the state's birth defects registry, University of North Carolina at Charlotte, and the North Carolina Department of Public Instruction will participate in all phases of the study. These individuals will be co-investigators and members of an Advisory Board. Results will improve understanding of short and long-term outcomes among children with OFC. This can enhance health and quality of life for children with these conditions and their families.