Barriers to Health Care for Children with Orofacial Clefts: A Systematic Literature Review and Recommendations for Research Priorities

Background: There is a growing interest in health services research of orofacial clefts (OFC) in the United States. The objective of this study is to summarize the empirical knowledge to date about barriers to health care for children with OFC. Methods: We completed a systematic literature review to identify articles on barriers to health care for children with OFC in the United States. Pubmed, Embase, CINAHL, and Medline were searched from their dates of origin through June 2018 using a combination of key terms related to access and barriers to health care. Case reports and studies of populations outside of the United States were excluded. Results: 4079 publications were identified using our search strategy. After a title and abstract review, 18 were included in our review as they met inclusion criteria. These studies examined health care costs, health insurance coverage, access to team care, geographic barriers, adequacy of training of community-based providers in providing services to children with OFC, and socioeconomic and demographic factors. The key findings indicate much higher health care costs for children with OFC than unaffected children early in childhood, racial/ethnic disparities in certain access measures, and inadequate insurance coverage, distance to teams, and inadequate training of community-based providers in OFC-specific services as potential areas of concern. Except for studies on health care costs, the evidence is largely based on relatively small and primarily descriptive studies. Conclusions: The extant literature documents high health care costs for OFC treatments and suggests inadequate insurance coverage, long distance to cleft teams, and racial/ethnic disparities as critical factors related to access. We discuss multiple future research priorities. Among these, understanding the impacts of variation between states in mandates for private insurance benefits and generosity in Medicaid coverage on access to care as well as effects of differences in provider reimbursements are particularly understudied areas that can be meaningful for policymaking aimed at improving access and health outcomes of children with OFC. Examining access throughout childhood and later in life and employing robust designs and population-representative data are also important research and methodological extensions of the current literature.