Decisional Capacity and Informed Consent in Fragile X Syndrome

Not Applicable

Completed

Conditions: Fragile X Syndrome (FXS)

Interventions: Other: Digital Informed Consent Tool
Other: Paper and Pencil Informed Consent

Registration Number: NCT02465931

Lead Sponsor: RTI International

Brief Summary: The Decisional Capacity and Informed Consent in Fragile X Syndrome (FXS) project is for the Eunice Kennedy Shriver National Institute of Child Health \& Human Development, 1R01HD071987-01A1, and will provide the first comprehensive description of decisional capacity of individuals with FXS; identify individual, family, and experiential factors associated with variability in decisional capacity; determine the validity of caregiver and expert ratings; and develop evidence-based guidelines for categorizing decisional capacity of individuals with FXS. The researchers will develop a methodologically rigorous and conceptually grounded decision aid using digital technology to enhance participation of individuals with FXS in the consent process.

Detailed Description: FXS is the most common inherited form of intellectual disability (ID). The typical male has an IQ (intelligence quotient) of 50 to 55, but involvement ranges from mild to severe. Females are generally less affected and have a broader range in function. Some have severe ID and adaptive impairments, but most have a normal or borderline intellectual ability. This wide range in cognitive skills leads to variable ability to make choices and live independently. Most research on individuals with FXS has been noninvasive, limited to parent surveys and psychological assessments. Studies such as these evoke few if any major ethical concerns, so the consent process typically involves a straightforward parental consent. Some studies have drawn blood, collected saliva, measured heart rate, or used neuroimaging, but until recently there has been a paucity of treatment research. This scenario has changed dramatically in the past several years. Advances in understanding the molecular basis of FXS have led to a new generation of treatments, and clinical trials are under way using a variety of compounds. The possibility of side effects and the potential for significant changes in behavior and ability elevate to a new level the importance of obtaining meaningful consent, not only from parents, but also from individuals with FXS. Researchers and Institutional Review Board (IRB) members need data to guide decisions about involving individuals with FXS in the consent process. Unfortunately, little is known about the extent to which individuals with FXS can be or are involved in decisions about research participation. This project will assess the range of decisional capacity in FXS, identify factors associated with individual differences in decisional capacity, validate a rating scale for categorizing decisional capacity, and develop a novel tablet-based decision aid technology to assist researchers and clinicians in maximizing decisional capacity and consent. This study will work toward achieving two overarching aims: 1) Characterize and explain individual differences in decisional capacity in FXS, 2) Design and evaluate the effect of a tablet-based decision aid on participation in the consent process for a hypothetical clinical trial. Ultimately, this study will provide important information about the nature of decisional capacity in FXS and validate a novel intervention to maximize successful participation in the consent process.

Recruitment & Eligibility

Status: COMPLETED

Sex: All

Target Recruitment: 152

Inclusion Criteria

Places in the first 3 categories of the MacCAT flip chart
Can provide informed consent using a standard practice

Exclusion Criteria

Not in the first 3 categories of the MacCAT flipchart
Not able to provide informed consent without significant modifications to consent procedures

Study & Design

Study Type: INTERVENTIONAL

Study Design: PARALLEL

Arm && Interventions

Group	Intervention	Description
Intervention condition	Digital Informed Consent Tool	Digital informed consent tool
Comparison Condition	Paper and Pencil Informed Consent	Paper and pencil informed consent

Primary Outcome Measures

Name	Time	Method
Decisional Capacity - Understanding Score	Day 1, immediately following presentation of the material in the intervention or comparison condition	MacArthur Decisional Capacity - understanding score, range 0 (no understanding) to 26 (perfect understanding) A summary score was calculated, ranging from 0-26. Scores were then translated into a percentage based on the number of questions answered. A score of 100% indicates that the participant answered each understanding question correctly on the first attempt.

Secondary Outcome Measures