Should we be studying family communication tools to improve cascade testing in genomics? A value-of-information analysis in Lynch syndrome

Abstract

Purpose: To estimate the potential value of family communication tools to improve cascade screening in Lynch syndrome and the expected value of additional research to reduce uncertainty in their effectiveness. Methods: We developed a decision tree and Markov model, which consists of four states: well, colorectal cancer (CRC) (year 1), CRC (years 2-5+) and death. A preliminary estimate of the effectiveness of a communication tool was based on a pilot RCT of a tool we previously developed, FamilyTalk. Clinical probabilities, utilities, and costs were derived from published literature and secondary databases. Value-of-information (VOI) analysis was used to estimate the expected value of additional evidence generated from future research. Results: The communication tool led to a difference in costs ranging from -$1,608 to $4,632 and a difference in quality-adjusted life-years (QALYs) from -0.26 to 0.83. The estimated value of future research specifically on the effectiveness of a communication tool was $52 million. In contrast, further research on reducing the uncertainties of adherence to or effectiveness of annual colonoscopy did not contribute to decreased decision uncertainty. Conclusion: A family communication tool to improve Lynch syndrome cascade screening of even moderate effectiveness likely provides good economic value, but further research is warranted.