Decision Making in Expanded Newborn Screening: The Case of Duchenne Muscular Dystrophy.

Newborn Screening Decision Making;Optional Newborn Screening;Duchenne Muscular Dystrophy Newborn Screening;Pediatrics;Public Health;Health Sciences;Health Behavior & Health Education

Lillie, Sarah ElizabethTarini, Beth A. ;

University of Michigan

关键词: Newborn Screening Decision Making;    Optional Newborn Screening;    Duchenne Muscular Dystrophy Newborn Screening;    Pediatrics;    Public Health;    Health Sciences;    Health Behavior & Health Education;   

Others  :  https://deepblue.lib.umich.edu/bitstream/handle/2027.42/98052/selillie_1.pdf?sequence=1&isAllowed=y

瑞士|英语

来源: The Illinois Digital Environment for Access to Learning and Scholarship

【 摘 要 】

Background. Newborn screening (NBS) is a mandatory public health program aimed at the early identification of babies with conditions that will benefit from early diagnosis and treatment. With increasing technology, some programs have offered optional NBS for diseases for which there is limited treatment efficiency data. This dissertation used Duchenne muscular dystrophy (DMD) as an exemplar to address whether variation in the presentation and structural characteristics of NBS influence decision making. Methods.In 3 randomized survey experiments using Internet samples, I explored characteristics of test structure and presentation that may motivate utilization of DMD NBS. The primary outcome variable was intent to utilize DMD NBS with additional outcome variables of attitudes towards DMD NBS. Results.Providing a context of mandatory NBS (either bundled or unbundled) influenced DMD NBS intent and attitudes towards DMD NBS. When participants were not given this broader context in which to place a specific optional NBS, they were more hesitant to choose testing. Neither the mode of results release, nor the overall test purpose guiding the release, were significant predictors when parents lacked any specific reason to believe their child was at risk. However, an interaction of DMD NBS purpose and perceived vulnerability showed that personal purpose increased DMD NBS intent when perceived vulnerability existed, but DMD NBS intent was relatively consistent regardless of perceived vulnerability when the test’s main purpose was research. Conclusions. New parents are increasingly being faced with optional NBS decisions, yet there is no consistent policy regarding optional NBS communication, in terms of the information included and the way this information is presented. The results suggest that future optional NBS programs should be careful to present testing information in a way that explains how a single optional NBS test fits into overall mandatory NBS. Additionally, health professionals should attend to parents’ perceptions of their child’s vulnerability, which appears to be a broader construct than simply family history of a specific disease. Increasing attention to the influence of such structural factors and individual differences on optional NBS decision making will become more and more important as NBS programs are likely to continue expanding.