, Volume 22, Issue 1, pp 108-117
Date: 27 Jun 2012

Design and Evaluation of a Decision Aid for Inviting Parents to Participate in a Fragile X Newborn Screening Pilot Study

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The major objectives of this project were to develop and evaluate a brochure to help parents make an informed decision about participation in a fragile X newborn screening study. We used an iterative development process that drew on principles of Informed Decision Making (IDM), stakeholder input, design expertise, and expert evaluation. A simulation study with 118 women examined response to the brochure. An independent review rated the brochure high on informational content, guidance, and values. Mothers took an average of 6.5 min to read it and scored an average of 91.1 % correct on a knowledge test. Most women rated the brochure as high quality and trustworthy. When asked to make a hypothetical decision about study participation, 61.9 % would agree to screening. Structural equation modeling showed that agreement to screening and decisional confidence were associated with perceived quality and trust in the brochure. Minority and white mothers did not differ in perceptions of quality or trust. We demonstrate the application of IDM in developing a study brochure. The brochure was highly rated by experts and consumers, met high standards for IDM, and achieved stated goals in a simulation study. The IDM provides a model for consent in research disclosing complicated genetic information of uncertain value.

Primary support for this study was provided by the National Newborn Screening and Genetics Resource Center, through a cooperative agreement between the Maternal and Child Health Bureau, Genetic Services Branch and the University of Texas Health Science Center at San Antonio, Department of Pediatrics, HRSA Grant # U32-MC00148. Partial support was also provided by the Eunice Kennedy Shriver National Institute for Child Health and Human Development (P30 HD003110-S1), the Ethical, Legal, and Social Implications Research Program/National Human Genome Research Institute (5P50HG004488), and the Centers for Disease Control in conjunction with the Association for Prevention Teaching and Research (APTR) (Cooperative Agreement No. U50/CCU300860, Project TS-1470). The authors express their appreciation to Debra Skinner for her comments on the manuscript and assistance with coding, and to colleagues at the University of California-Davis and Rush University Medical Center for suggestions on the brochure.