Decisional Capacity and Informed Consent in Fragile X Syndrome

• ClinicalTrials.gov Identifier: NCT02465931
• Recruitment Status: Completed
• First Posted: June 9, 2015
• Results First Posted: December 6, 2018
• Last Update Posted: March 7, 2019
• Sponsor: RTI International
• Collaborator: Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD)
• Information provided by (Responsible Party): RTI International

Study Description

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.

Condition or disease	Intervention/treatment	Phase
Fragile X Syndrome (FXS)	Other: Paper and Pencil Informed Consent; Other: Digital Informed Consent Tool	Not Applicable

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.

Study Design

• Study Type: Interventional (Clinical Trial)
• Actual Enrollment: 152 participants
• Allocation: Randomized
• Intervention Model: Parallel Assignment
• Masking: None (Open Label)
• Primary Purpose: Screening
• Official Title: Decisional Capacity and Informed Consent in Fragile X Syndrome
• Actual Study Start Date: March 14, 2016
• Actual Primary Completion Date: March 21, 2017
• Actual Study Completion Date: March 21, 2017

Arms and Interventions

Arm	Intervention/treatment
Comparison Condition; Paper and pencil informed consent	Other: 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.
Experimental: Intervention condition; Digital informed consent tool	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.

Outcome Measures

Primary Outcome Measures :

1. Decisional Capacity - Understanding Score [ Time Frame: 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.

Eligibility Criteria

• Ages Eligible for Study: 12 Years to 40 Years (Child, Adult)
• Sexes Eligible for Study: All
• Accepts Healthy Volunteers: No

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

Contacts and Locations

Locations

United States, North Carolina

RTI International

Research Triangle Park, North Carolina, United States, 27709

Sponsors and Collaborators

RTI International

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

Investigators

• Study Director: Robert Furberg, PhD; RTI International

  Study Documents (Full-Text)

Documents provided by RTI International:

Study Protocol and Statistical Analysis Plan  [PDF] June 11, 2018

More Information

Publications automatically indexed to this study by ClinicalTrials.gov Identifier (NCT Number):

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.

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.

• Responsible Party: RTI International
• ClinicalTrials.gov Identifier: NCT02465931
• Other Study ID Numbers: FXS; 1R01HD071987-01A1 ( U.S. NIH Grant/Contract )
• First Posted: June 9, 2015
• Results First Posted: December 6, 2018
• Last Update Posted: March 7, 2019
• Last Verified: February 2019

Individual Participant Data (IPD) Sharing Statement:

• Plan to Share IPD: No

Keywords provided by RTI International:

Informed Consent; Fragile X Syndrome; Tablets; Decisional Capacity

Additional relevant MeSH terms:

Fragile X Syndrome; Syndrome; Disease; Pathologic Processes; Mental Retardation, X-Linked; Intellectual Disability; Neurobehavioral Manifestations; Neurologic Manifestations; Nervous System Diseases; Sex Chromosome Disorders; Chromosome Disorders; Congenital Abnormalities; Genetic Diseases, Inborn; Genetic Diseases, X-Linked; Heredodegenerative Disorders, Nervous System