Estimating Preferences for Complex Health Technologies: Lessons Learned and Implications for Personalized Medicine

• Published in: Value in health Vol. 20; no. 1; pp. 32 - 39
• Main Authors: Marshall, Deborah A., PhD, Gonzalez, Juan Marcos, PhD, MacDonald, Karen V., MPH, Johnson, F. Reed, PhD
• Format: Journal Article
• Language: English
• Published: United States Elsevier Inc 2017; Elsevier Science Ltd
• Subjects: Biomedical research; Biomedical Technology; Biotechnology; Choice Behavior; Contingent valuation; Decision Support Techniques; discrete-choice experiment; Endogenous; Evidence-based medicine; genetic testing; Genetic Testing - economics; Genomes; Genomics; Health care; Health problems; Humans; Internal Medicine; patient acceptance of health care; Patient Acceptance of Health Care - psychology; patient preference; Patient Preference - psychology; personalized medicine; Precision medicine; Precision Medicine - economics; Research Design; Severity of Illness Index; Uncertainty; Usefulness; Variants; patient preference; discrete-choice experiment; choice behavior; genetic testing; patient acceptance of health care; personalized medicine
• ISSN: 1098-3015; 1524-4733
• DOI: 10.1016/j.jval.2016.08.737

Abstract We examine key study design challenges of using stated-preference methods to estimate the value of whole-genome sequencing (WGS) as a specific example of genomic testing. Assessing the value of WGS is complex because WGS provides multiple findings, some of which can be incidental in nature and unrelated to the specific health concerns that motivated the test. In addition, WGS results can include actionable findings (variants considered to be clinically useful and can be acted on), findings for which evidence for best clinical action is not available (variants considered clinically valid but do not meet as high of a standard for clinical usefulness), and findings of unknown significance. We consider three key challenges encountered in designing our national study on the value of WGS—layers of uncertainty, potential downstream consequences with endogenous aspects, and both positive and negative utility associated with testing information—and potential solutions as strategies to address these challenges. We conceptualized the decision to acquire WGS information as a series of sequential choices that are resolved separately. To determine the value of WGS information at the initial decision to undergo WGS, we used contingent valuation questions, and to elicit respondent preferences for reducing risks of health problems and the consequences of taking the steps to reduce these risks, we used a discrete-choice experiment. We conclude by considering the implications for evaluating the value of other complex health technologies that involve multiple forms of uncertainty.