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NIH Sep 15, 2018 | R01
Unresolved Issues in Newborn Screening: Quantifying the Harms of a False Positive Result
Institution: Children's Hospital
FOA Number: PA-16-160
Abstract
While newborn screening (NBS) programs have saved the lives of thousands of children with inherited disorders, the future success of this vital public health program depends on the ability to accurately assess and balance the benefits and harms of screening. The ongoing development of new testing technologies increases the number of disorders screened, which generates more false positive NBS results. Despite decades of research, the scope and magnitude of harms from false positive NBS results remain poorly understood. Failure to resolve this poor understanding has significant policy, ethical, and research implications. From a policy perspective, it undermines the requisite evaluation of benefits and harms that accompanies decisions about whether to add a disorder to a state NBS panel. From an ethical perspective, it places children and their families at risk for adverse outcomes from a mandatory public health program. From a research perspective, it interferes with the design of effective interventions to mitigate these harms. Therefore, the objective of this study is to comprehensively examine the scope, magnitude, and risk factors for harms to both children who receive false positive NBS results and their families. This study objective will be accomplished through a prospective multistate cohort study of children undergoing NBS and their parents with the following specific aims: (1) To determine the scope and magnitude of harms due to false positive NBS results; (2) To identify parental and child factors associated with harms due to false positive NBS results; and (3) To develop a primary care provider (PCP)“parent communication aid to improve PCP communication with parents of children who receive false positive NBS results. The proposed project will provide the most comprehensive data to-date on post-screening harms for children with false positive NBS results. These data are critical to the success of future efforts to improve and expand NBS in the US.
FUNDING AGENCY:
Funder:
NIHInstitute:
EUNICE KENNEDY SHRIVER NATIONAL INSTITUTE OF CHILD HEALTH & HUMAN DEVELOPMENTFunding Type:
R01Project Number:
R01HD095068Start Date:
Sep 15, 2018End Date:
Jun 30, 2023PROJECT TERMS:
adverse outcome, Behavioral, Bioethics, Biometry, Birth Order, care providers, Child, Chronic, Cohort Studies, Communication, communication aid, Data, design, Development, Disease, Distress, Economics, effective intervention, Effectiveness, Ensure, Equilibrium, Ethics, Evaluation, Evidence based intervention, experience, Failure, Family, follow-up, Future, Genetic Counseling, Goals, Health, Health Services, Hereditary Disease, Impairment, implementation research, improved, innovation, Interdisciplinary Study, Length, Link, Medical, Mental disorders, Methodology, National Institute of Child Health and Human Development, Neonatal Screening, Outcome, Parents, Policies, programs, prospective, Psychology, psychosocial, Public Health, public policy, Recording of previous events, Research, Research Personnel, Risk, Risk Factors, screening, screening panel, screening program, Screening Result, standardize measure, study population, success, Technology, Testing, Time, Translational Research, United States National Institutes of Health