title: 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 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.
authors.affiliation: RTI International, Research Triangle Park, Durham, NC, USA dbailey@rti.org.
authors.forename: Donald B
authors.initials: DB
authors.lastname: Bailey
authors.affiliation: RTI International, Research Triangle Park, Durham, NC, USA.
authors.forename: Melissa
authors.initials: M
authors.lastname: Raspa
authors.affiliation: RTI International, Research Triangle Park, Durham, NC, USA.
authors.forename: Anne
authors.initials: A
authors.lastname: Wheeler
authors.affiliation: RTI International, Research Triangle Park, Durham, NC, USA.
authors.forename: Anne
authors.initials: A
authors.lastname: Edwards
authors.affiliation: RTI International, Research Triangle Park, Durham, NC, USA.
authors.forename: Ellen
authors.initials: E
authors.lastname: Bishop
authors.affiliation: RTI International, Research Triangle Park, Durham, NC, USA.
authors.forename: Carla
authors.initials: C
authors.lastname: Bann
authors.affiliation: University of North Carolina, Chapel Hill, NC, USA.
authors.forename: David
authors.initials: D
authors.lastname: Borasky
authors.affiliation: Columbia University, New York, NY, USA.
authors.forename: Paul S
authors.initials: PS
authors.lastname: Appelbaum
mesh_terms: D000328:Adult; D002986:Clinical Trials as Topic / Q000941:ethics*; D032882:Comprehension; D003657:Decision Making; D006233:Persons with Disabilities; D032984:Ethics, Research; D005190:Family; D005260:Female; D005600:Fragile X Syndrome / Q000188:drug therapy; D006801:Humans; D007258:Informed Consent; D008297:Male; D008875:Middle Aged; D010290:Parents; D010465:Perception; D035842:Research Subjects; D011795:Surveys and Questionnaires
keywords: clinical trials; decisional capacity; fragile X syndrome; informed consent