Parent Ratings of Ability to Consent for Clinical Trials in Fragile X Syndrome

Abstract

Advances in understanding neurobiology and intellectual disabilities have led to clinical trials testing new medications. This study assessed parents’ perceptions of the ability of their son or daughter with fragile X syndrome (FXS), an inherited form of intellectual disability, to participate in the consent process for clinical trials. Four hundred and twenty-two families participated in a survey that included six items assessing various aspects of the ability to provide consent. A rank ordering of decisional tasks was found. The easiest task was to understand that the medication was different from his or her medical treatment; the most difficult was the ability to understand and weigh the potential benefits and risks of study participation. Factor analysis suggested that despite the range in difficulty, the six items were best summarized by a single decisional ability score. Parents of 29% of males reported that their son was not at all capable of participating, but the remainder exhibited a range of decisional skills. Factors associated with this variability include age, and parents’ willingness to enroll their child in clinical trials. We conclude that many individuals with FXS appear to be able to participate at some level in the consent or assent process, but will likely need individualized support to maximize effective participation.

A Randomized Controlled Trial of an Electronic Informed Consent Process

“…Identifying new mechanisms to improve research participant comprehension during the process is necessary to promote the ethical conduct of research. However, along with other research evidence, this study demonstrates that different formats of informed consent may not be applicable to all research contexts (Bailey et al, 2014). For example, in this study, we found that for 6 of the 14 items on the survey, participants in the simplified paper-based consent group had higher mean scores than those in the electronic informed consent group.…”

A Digital Decision Support Tool to Enhance Decisional Capacity for Clinical Trial Consent: Design and Development (Preprint)

“…Males diagnosed with FXS typically have impairment ranging from mild to severe; females are generally less impaired [12]. This wide range of cognitive skills among those with FXS can result in variable decisional capacity and the ability to make choices [13].…”

“…To date, most research on individuals with FXS has been noninvasive, limited to parent surveys and secondary assessment of clinical data [13,14]. Studies such as these typically involve straightforward consent/assent processes or parental consent.…”

“…Studies such as these typically involve straightforward consent/assent processes or parental consent. However, with advances in understanding the underlying mechanisms of FXS, there has been an increase in the number of clinical trials available for individuals with FXS [13,15]. Decisions related to enrollment in treatment trials are now more complex than in the past, thus researchers are compelled to consider how best to support decision-making for individuals who present with a range of decisional capacity.…”

“…The few studies that have examined ways to support individuals with ID in the informed consent process have found that the presentation of information is important, given that language skills, memory, and previous decision-making all have an impact on ability to consent [16]. Due to the wide range in decisional capacity, those with ID could participate in the consent process, but many authors encourage that participation should be determined and supported on an individual case-by-case basis [13,16,17].…”