Decisional Capacity and Informed Consent in Fragile X Syndrome

NCT ID: NCT02465931

Last Updated: 2019-03-07

Basic Information

• Recruitment Status: COMPLETED
• Clinical Phase: NA
• Total Enrollment: 152 participants
• Study Classification: INTERVENTIONAL
• Study Start Date: 2016-03-14
• Study Completion Date: 2017-03-21

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.

Conditions

Fragile X Syndrome (FXS)

Study Design

• Allocation Method: RANDOMIZED
• Intervention Model: PARALLEL
• Primary Study Purpose: SCREENING
• Blinding Strategy: NONE

Study Groups

Comparison Condition

Paper and pencil informed consent

Group Type: OTHER

Paper and Pencil Informed Consent

Intervention Type: OTHER

Paper-based informed consent form will be sent to participant and family before data collection visit. They will be able to review as many times as they wish before visit. During visits, simplified overview of informed consent form will be provided in person just once. For MacArthur Competence Assessment Tool (MacCAT) Questions, All questions will be asked after the disclosure information has been presented. Questions will have the same wording as experimental condition. Procedures will mimic MacCAT/flipchart (incorrect or partial credit will be given opportunity to answer question again after disclosure information is repeated). There will be multiple choice options rather than open-ended. Finally, data collection will be done through paper and pencil.

Intervention condition

Digital informed consent tool

Group Type: EXPERIMENTAL

Digital Informed Consent Tool

Intervention Type: OTHER

Paper informed consent form will be sent to participant and family before data collection visit. They will be able to review as many times as they wish before visit. During visits, the participant can go through the tablet-based tool up to 3 times. All questions will be embedded within the vignettes/presentation of disclosure information. Question will use simplified wording, similar to flipchart. Procedures will mimic MacArthur Competence Assessment Tool (MacCAT) /flipchart (incorrect or partial credit will be given opportunity to answer question again after disclosure information is repeated). Multiple choice options will be utilized rather than open-ended. Finally, response data stored within tool and exported to dataset for analysis.

Interventions

Paper and Pencil Informed Consent

Paper-based informed consent form will be sent to participant and family before data collection visit. They will be able to review as many times as they wish before visit. During visits, simplified overview of informed consent form will be provided in person just once. For MacArthur Competence Assessment Tool (MacCAT) Questions, All questions will be asked after the disclosure information has been presented. Questions will have the same wording as experimental condition. Procedures will mimic MacCAT/flipchart (incorrect or partial credit will be given opportunity to answer question again after disclosure information is repeated). There will be multiple choice options rather than open-ended. Finally, data collection will be done through paper and pencil.

Intervention Type: OTHER

Digital Informed Consent Tool

Paper informed consent form will be sent to participant and family before data collection visit. They will be able to review as many times as they wish before visit. During visits, the participant can go through the tablet-based tool up to 3 times. All questions will be embedded within the vignettes/presentation of disclosure information. Question will use simplified wording, similar to flipchart. Procedures will mimic MacArthur Competence Assessment Tool (MacCAT) /flipchart (incorrect or partial credit will be given opportunity to answer question again after disclosure information is repeated). Multiple choice options will be utilized rather than open-ended. Finally, response data stored within tool and exported to dataset for analysis.

Intervention Type: OTHER

Eligibility Criteria

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

• Minimum Eligible Age: 12 Years
• Maximum Eligible Age: 40 Years
• Eligible Sex: ALL
• Accepts Healthy Volunteers: No

Sponsors

Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD)

NIH

Sponsor Role: collaborator

RTI International

OTHER

Sponsor Role: lead

Responsible Party

Responsibility Role: SPONSOR

Principal Investigators

Robert Furberg, PhD

Role: STUDY_DIRECTOR

RTI International

Locations

RTI International

Research Triangle Park, North Carolina, United States

Countries

United States

References

Furberg RD, Raspa M, Wheeler AC, McCormack LA, Bailey DB. A Digital Health App to Assess Decisional Capacity to Provide Informed Consent: Protocol for a Randomized Controlled Trial. JMIR Res Protoc. 2018 Nov 19;7(11):e10360. doi: 10.2196/10360.

Reference Type: DERIVED

PMID: 30455171 (View on PubMed)

Furberg RD, Ortiz AM, Moultrie RR, Raspa M, Wheeler AC, McCormack LA, Bailey DB Jr. A Digital Decision Support Tool to Enhance Decisional Capacity for Clinical Trial Consent: Design and Development. JMIR Res Protoc. 2018 Jun 6;7(6):e10525. doi: 10.2196/10525.

Reference Type: DERIVED

PMID: 29875084 (View on PubMed)

Provided Documents

Document Type: Study Protocol and Statistical Analysis Plan

View Document

Other Identifiers

1R01HD071987-01A1

Identifier Type: NIH

Identifier Source: secondary_id

View Link

FXS

Identifier Type: -

Identifier Source: org_study_id