A pilot assessment of parental practices and attitudes regarding risk disclosure and clinical research involving children in Huntington disease families

Abstract

PurposeTo characterize parental practices of informing children of risk for Huntington disease (HD), and to understand the attitudes of parents concerning childhood participation in HD research. MethodsAn anonymous Internet survey was accessed by individuals of HD families. The survey probed for data regarding individual risk for HD, as well as when or if children had been informed of the disease. Respondents expressed their attitudes concerning childhood participation in HD clinical research. ResultsTwo hundred forty-nine individuals responded (∼80% female), and 84% had never participated in an HD clinical trial. Seventy-five percent of respondents were parents; nearly two thirds of them had provided some information about HD to their children. There was overwhelming support for affected, at-risk, and unaffected adults in terms of HD research participation, but there was a statistically significant disparity by gene status, with gene negative and symptomatic gene positive adults being more inclined to participate than at-risk or asymptomatic/gene positive adults. More than 50% of respondents supported childhood participation, but typically in late adolescence (15–18 years). Gene negative and symptomatic adults were statistically more likely to agree with childhood inclusion than at-risk or asymptomatic/gene positive adults. ConclusionThese results serve as pilot data for further investigations to address childhood participation in HD research. In addition, these findings will inform ongoing studies as to appropriate practices to undertake to include minors.