Research attitudes in families of individuals with Down syndrome: importance for clinical trials

Abstract

Background Individuals with Down syndrome (DS) are increasingly eligible for clinical trial intervention, particularly for the treatment or prevention of Alzheimer disease (AD). Yet, little is known about research attitudes that may contribute to decisions regarding clinical trial enrollment for people with DS, a gap which is addressed in the current study. Methods The Research Attitudes Questionnaire (RAQ) is a brief validated instrument that measures cultural and social factors which influence clinical trial enrollment decisions in the general population. Applied herein to a cohort of 1002 families who have an individual with DS, this survey was carried out through a national registry (DS-Connect). In addition to the RAQ, demographic data were collected. Results The response rate to the survey was 49.9%. Respondents were asked to complete demographic information and to respond to the 7 question RAQ. The scores were stratified by a cut point assigned a priori into those more favorable toward research participation vs. those less favorably inclined. Within this sample, nearly 95% self-identified as the primary caretaker for the individual with DS. The RAQ score analyses generally indicated favorable respondent views toward research with particularly high favorability ratings from respondents who had previously participated in research and from those who were older (P = .01 to .001). Conclusions This is one of the first formal studies to evaluate research attitudes among relatives of individuals with DS and shows the feasibility of using this approach to answer important questions that will guide trialists developing treatments for AD in DS. Future research will require broadening the racial and ethnic mix of respondents and the role that a standardized assessment of research attitudes will have for clinical trial participation.

Comparing research attitudes in Down syndrome and non‐Down syndrome research decision‐makers

“…In item‐level analyses, DS decision‐makers were more favorably disposed toward “responsibility for helping others through volunteering for medical research.” In our previous study, favorable attitudes toward research participation were also noted. 13 In children with DS under the age of 18 years, 72% of their parents expressed willingness to participate in research, although only 36% had actually enrolled in a clinical trial. Barriers to research participation for people with DS have been reviewed and include benefit‐risk assessment, time commitment, access to results, and the “power distance” between the researchers and families.…”

“…RAQ scores have been previously shown to be associated with willingness to participate in research, compliance with study protocols, and study completion. 9 , 10 , 11 , 12 In a previous study, we showed the feasibility of using the RAQ in over 1000 family members for individuals with DS, 13 but we still do not understand how individual items on the RAQ compare to those in the general population. In the present report, we explored differences in RAQ scores between healthcare and research enrollment decision‐makers for individuals with DS and those from a more general population, both of which come from research‐friendly registries in the United States.…”

Research Attitude and Interest among Cancer Survivors with or without Cognitive Impairment

“…A summary score, ranging from 7 to 35, was calculated and weighted against the number of missing RAQ responses for each participant, with higher scores representing more positive research attitudes. Previous studies have demonstrated a relationship between the RAQ score and willingness to participate in research, as well as the likelihood of completion of clinical trials [15][16][17]. (2).…”

“…A summary score, ranging from 7 to 35, was calculated and weighted against the number of missing RAQ responses for each participant, with higher scores representing more positive research attitudes. Previous studies have demonstrated a relationship between the RAQ score and willingness to participate in research, as well as the likelihood of completion of clinical trials [ 15 , 16 , 17 ]. Willingness to participate in research activities: Participants were asked about their interest in being notified about studies (yes/no) which investigate approved medication, investigational medicine, diet or lifestyle alteration, blood draws, cognitive testing, magnetic resonance imaging (MRI), positron emission tomography (PET), lumbar puncture, autopsy, and on-site/at-home blood draws for genetic testing and biomarker quantification [ 10 ].…”

The National Institutes of Health INvestigation of Co‐occurring conditions across the Lifespan to Understand Down syndromE (INCLUDE) Project: Accelerating research discoveries for people with Down syndrome across the lifespan

“…Seven are examining intervention strategies for obstructive sleep apnea, three are addressing aging and AD risk, one is evaluating immune modulatory treatments for autoimmune skin A list of studies that are currently recruiting through the registry can be found on the DS-Connect website. Among the projects supported by the registry are a survey of dietary supplement use in children with DS (Lewanda et al, 2018), a survey of parents utilizing a validated tool to assess measures of global health in their children with DS (S. L. Santoro, Haugen, et al, 2023), application of the Research Attitudes Questionnaire to over 1000 families in the DS-Connect Registry (Lott et al, 2022), the comfort of caregivers with the transition to adult care for their children with DS (VanZant & McCormick, 2021), and a survey of preventive and gynecologic health care uptake by adult women with DS (Smith et al, 2020). One of the most significant events sponsored by the…”