Decisional Capacity and Informed Consent in Fragile X Syndrome
Condition: Fragile X Syndrome (FXS)
MeSH terms: Fragile X Syndrome; Syndrome
Study type: Interventional

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.
    

Intervention name(s): Paper and Pencil Informed Consent; Digital Informed Consent Tool
Intervention type(s): Other; Other
Intervention description(s): 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.; 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.
Allocation: Randomized
Intervention model: Parallel Assignment
Masking: None (Open Label)
Primary purpose: Screening
Keywords: Informed Consent; Fragile X Syndrome; Tablets; Decisional Capacity