Attitudes Towards Medical Research Participation Among Those With Down Syndrome and Their Caregivers

Abstract

Background:

In the United States, recent increases in funding and support for research related to Down syndrome have led to increased opportunities for research participation. It is critical to understand how individuals with Down syndrome and their carers view participation in research, as these individuals are major stakeholders in the research process. The goal of this study was to collect feedback regarding attitudes towards research directly from individuals with Down syndrome, as well as their caregivers.

Methods:

A cross-sectional online survey of individuals with Down syndrome and caregivers assessed attitudes towards research participation, including feedback regarding facilitators and barriers to participation, willingness to participate in specific procedures, and how the research process might best serve participants.

Results:

Respondents included 46 persons with Down syndrome (age 16–61 years; 48% male, 52% female) and 216 caregivers (age 29–88 years; 9% male, 91% female). The potential to improve the lives of people with Down syndrome emerged as a strong incentive for research participation among both caregivers (96%) and respondents with Down syndrome (82%). Compared to caregivers, respondents with Down syndrome were more open to participating in research that included the risk of pain or the need to do something difficult. Compared to respondents with Down syndrome, caregivers responded more positively to being able to stay with the focus person during research procedures. Response patterns showed few relationships to age, sex, or previous research participation.

Conclusion:

The responses of people with Down syndrome and caregivers showed some similarities, with notable differences highlighting the importance of respecting the autonomy of the participant. Specific recommendations related to the research process include the need to give participants understandable, thorough information related to research procedures and the importance of consistent communication.

Introduction

Typically, ethical considerations are at the forefront of research planning. At most institutions, research with human participants requires detailed review of the planned procedures, weighing risks undertaken by research participants against benefits to participants and the broader community. As major stakeholders, it is increasingly common for researchers to include representatives of the research population in both the initial development of research protocols and as members of the research team. Published investigations of attitudes towards research participation within a specific population can supplement this process, allowing researchers gain a broad understanding of topics of concern and providing a starting point for inclusive research.

There is limited research investigating attitudes towards research participation among people with intellectual disabilities and their families and carers (Kalb et al., 2019; McDonald et al., 2013; Nicholson et al., 2013; Scotti et al., 2012). Both caregivers (Kalb et al., 2019; Scotti et al., 2012) and individuals with intellectual disabilities (Haas et al., 2016; McDonald et al., 2013) are generally positive towards research participation, with benefit to the larger community being a strong motivator (Haas et al., 2016). Until recently, there were no such studies focused specifically on individuals with the genetic condition Down syndrome. In the United States, funding for Down syndrome–related research has increased with the implementation of the National Institutes of Health (NIH) Investigation of Co-occurring conditions across the Lifespan to Understand Down syndrome (INCLUDE) Project (INCLUDE Project, 2024), resulting in a wider variety of research topics and a need for larger numbers of research participants. An understanding of attitudes towards research participation among those with Down syndrome and their carers can improve the research experience for participants and lead to more effective recruitment and retention efforts.

This need was recognized by two recent surveys of carers of individuals with Down syndrome (Kyprianou et al., 2023; White et al., 2022). White et al. (2022) focused on parents with children under 18, asking how they learn about Down syndrome–related research, their willingness to participate, attitudes towards research, and interest in research topics. Kyprianou et al. (2023) surveyed carers of adults with Down syndrome to determine the likelihood of participation in different types of research. Both studies found a high degree of interest in research participation, particularly for non-invasive procedures. White et al. (2022) found that 92% of parents wanted “more treatment options related to drugs, therapies and interventions” (p. 183) for people with Down syndrome. At the same time, both studies found low willingness to participate in clinical trials of drugs or devices, highlighting the need for identification and mitigation of barriers to research participation (Kyprianou et al., 2023; White et al., 2022).

The current work expands on these findings and addresses a gap in knowledge by surveying attitudes towards research participation in those who are most directly impacted by research, individuals who have Down syndrome. For this study, both individuals with Down syndrome and caregivers responded to a cross-sectional online survey aiming to understand the incentives, disincentives, facilitators, and barriers to research participation, the willingness to participate in specific research procedures, how participants learn about research, and what might improve the research process for participants.

Methods

Survey development

A literature search identified research examining attitudes towards research participation in the general population and in populations that may face similar issues as people with Down syndrome and their carers (Haas et al., 2016; Kalb et al., 2019; Kim et al., 2009; McDonald et al., 2013; Quinn & Menon, 2020). For example, issues surrounding consent and assent, full understanding of research procedures, and balancing the interests of individuals and their carers can also impact participants with intellectual disabilities or dementia. These studies were used to identify potential topic areas and, in conjunction with literature on survey design (Dillman & Smyth, 2007; Sinkowitz-Cochran, 2013), to construct an initial set of questions. Questions about research attitudes were broadly categorized as:

1. Practical considerations (e.g. time commitment, location, benefits to the participants and community);

2. Welfare of individuals with Down syndrome (e.g. sensitivity to their wishes and concern for their well-being);

3. Research team and procedures (e.g. recruitment process, communication, coordination, reputation of researchers).

A draft survey was distributed to multiple stakeholders who provided feedback on content, wording, and readability through an iterative process via email and phone. Reviewers included educators, clinicians, and researchers who focus on intellectual disabilities or Down syndrome; the staff of an advocacy organization for individuals with Down syndrome; multiple individuals with Down syndrome and their families; and a psychologist who specializes in survey design.

Participants

The survey was developed to collect data from two categories of respondents: people with Down syndrome responding on their own and caregivers of people with Down syndrome. Caregiver respondents were defined as anyone involved in decision-making for a person with Down syndrome. The recruitment goal was 50 people with Down syndrome and 250 caregivers.

The survey landing page included a question to differentiate respondents, and branching logic was used to ensure that respondents were presented with relevant questions (Fig. 1).

Figure 1.

Survey flow. Branching logic was used to present respondents with relevant questions. Caregivers (CGs) completed the survey questions shown on the left. Self-advocates (SAs) completed the survey questions shown on the right. Double arrows indicate the survey flow when self-advocates completed the survey with their caregivers. FP, focus person.

1. For clarity, this analysis refers to people with Down syndrome who responded to the survey questions as “self-advocates.” The survey provided two ways for self-advocates to respond:

   1. Respondents could start the survey on their own, identifying as a person with Down syndrome. Although the survey did not include a formal consent process, for ethical purposes, self-advocates starting the survey on their own were required to state that they functioned as their own legal guardians. After identifying as a self-advocate, the survey asked the respondent to confirm that they are their own legal guardian, including an explanation of the term. In the case of a “yes” response, the respondent would proceed to the remainder of the survey (Fig. 1, right). A response of “no” or “I’m not sure” returned a page encouraging the respondent to talk to their caregiver about the survey.
   2. Respondents with Down syndrome who had a legal guardian were able to take the survey with their legal guardian (see “self-advocate + caregiver” below).

2. Caregivers. The survey asked that those responding as caregivers be involved in the decision-making process for a person with Down syndrome. Throughout the survey, the person with Down syndrome associated with the caregiver was referred to as the “focus person.” The caregiver could take the survey on their own, answering questions in reference to the focus person (Fig. 1, left). If the caregiver indicated that they were the legal guardian, they could allow their focus person to respond to the self-advocate portion of the survey (self-advocate + caregiver). In this case, the self-advocate questions were presented first (Fig. 1, double arrows). The respondent was notified at the completion of the self-advocate section, with the remainder of the survey asking questions of the caregiver only.

Survey content

The survey included three sections:

1. Demographics (age, sex, race, education, daily living skills)

2. Previous research participation

3. Attitudes towards research participation

Respondents were also asked how they receive and share information about Down syndrome generally and were provided a free response box to add comments.

After completing the demographics section, both self-advocates and caregivers were presented with a description of research. This survey focused primary on health and medical research participation, which was reflected in the description of research. This educational section was presented before questions about research participation and attitudes to ensure that all respondents had a similar understanding of the term “research” in the context of the survey.

Respondents who indicated that they had previously participated in research were asked about their participation in specific research procedures. Based on stakeholder feedback, self-advocates were presented with a slightly shorter list of previous research procedures to guard against fatigue. All participants were asked about attitudes towards research participation. In the research attitudes section, respondents were presented with a statement and used a Likert scale to indicate their response. Statements were presented in the form of both incentives and disincentives. The full text of survey questions related to previous research participation and research attitudes can be found in the supplementary materials under “Select survey components.”

In cases where a self-advocate responded to questions with a caregiver, the caregiver was not asked demographic questions about their focus person, as that information was captured in the self-advocate response section. However, the caregiver did complete questions related to their feelings about research participation involving the focus person. Consequentially, on select questions we were able to compare self-advocate responses directly to the responses given by their caregiver.

Survey administration

The survey was administered online from January 2022 through October 2023 through a public survey link using REDCap electronic data capture tools hosted at Cleveland Clinic (Harris et al., 2009). The survey was designed to allow respondents to remain anonymous, and reCAPTCHA technology was implemented in REDCap to lessen the possibility of automated responses. No compensation was given to respondents. All instruments and procedures were in accord with Declaration of Helsinki guidelines and were approved by the Cleveland Clinic Institutional Review Board (Federalwide Assurance 00005367). Because the survey did not collect identifying information and represented low risk, participants did not undergo a formal consent process. Rather, the first few screens presented information about risks and benefits, privacy, time commitment, and contact information for the researchers. To continue, participants had to indicate that they agreed to take the survey.

Recruitment

Multiple methods of recruitment were used. Regional Down syndrome advocacy organizations were asked to disseminate information about the survey via newsletter or social media, and researchers passed out informational fliers at organization events. The Cleveland Clinic patient database was used to identify current patients with a diagnosis of Down syndrome, and an informational letter about the survey was sent via mail or electronic communication (if available). Finally, the NIH-supported Down syndrome registry DS-Connect (dsconnect.nih.gov) was used to inform potential participants about the survey via email, with separate emails sent to caregivers and self-advocates.

Data analysis

The full dataset was inspected to identify incomplete responses or unusual response patterns. Respondents who provided no data or only demographic data were removed from further analysis. Group comparisons for continuous variables such as age and education were conducted using unpaired t-tests. Categorical variables, including responses to individual survey questions, were evaluated using χ² tests. The likelihood of previous research participation was related to demographic variables such as age and education. The supplementary material includes a comparison of self-advocate and caregiver responses to similar questions.

Results

Respondents

A total of 249 caregivers agreed to take the survey. Caregivers who provided no additional data or only basic demographic data were excluded from the analysis, resulting in 216 responses from caregivers. Fifty-five caregivers confirmed that they served as legal guardian to a person with Down syndrome and agreed to that person completing the self-advocate portion of the survey. In addition, four people with Down syndrome indicated that they were their own legal guardians and completed the survey. After accounting for self-advocates who provided no additional data or only basic demographic data, 46 self-advocates were included in the final analysis. See the supplemental material for additional details.

Demographics

Although we captured a large range of caregiver ages, our sample was homogenous in some respects (Table 1). Caregivers were predominately female (with 74% identifying as the mother of the focus person), Caucasian, and notably well-educated, with mean education of approximately 16 years, indicating a 4-year college degree (mean years 16.7 ± 2.49, maximum value set to 20 years). Most caregivers were also legal guardians (89%).

Table 1.

Survey respondent demographics

Demographic variable	Survey respondents
	SA (n = 46)	FP (n = 174)	CG (n = 213)*
Sex, %
Male	47.8	51.1	9.4
Female	52.2	48.9	90.6
Age, years
Mean (SD)	30.8 (10.8)	19.3 (16.1)	54.8 (13.3)
Minimum	16	0.33	29
Maximum	61	64	88
Race
African American	0	6	6
Asian	0	6	3
Caucasian	45	153	196
Native American	0	2	2
Other	1	5	5
Caregiver type
	n	% total
Mother	180	83.3
Father	20	9.3
Sibling	8	3.7
Other relative	7	3.2
Provider	0	0
Other	1	0.46

^*Three caregivers were missing demographic data but completed questions about research.

SA, self-advocate; FP, focus person; CG, caregiver; SD, standard deviation.

The age of self-advocate respondents spanned adulthood, and the sample showed a more even sex distribution than was seen for caregivers; however, the sample again lacked racial diversity (Table 1). All caregivers who did not respond with a self-advocate (n = 174) provided demographic information for their focus person (Table 1). The inclusion of children in the focus person group resulted in a lower overall age compared to self-advocates (p < 0.001), although the groups showed similar sex (p = 0.574) and racial (p = 0.389) distributions. See the supplemental material for additional characterization and comparison of the self-advocate and focus person samples.

Previous research participation

Table 2 shows the rates of previous research participation among respondents. The high rate of previous research participation among caregivers can be partially accounted for by those who had previously only completed questionnaires or surveys. Compared to caregivers who had not participated in any research, those who had participated had a higher level of education (p< 0.001). Previous research participation was not associated with caregiver age (p = 0.325). Caregivers with any previous research participation were more likely to report that their focus person had engaged in research (p < 0.001).

Table 2.

Participation in specific research procedures among survey respondents who participated in previous research

Research procedure	“Yes” respondents for each procedure, %
	SA (n = 28)	FP (n = 116)	CG (n = 163)
New medication	7.1	11.2	6.1
Changed behaviour	-	9.5	6.1
Kept track of behaviour	-	22.4	19.0
Blood draw	71.4	33.6	23.9
Medical examination	-	30.2	17.8
Non-invasive procedure	-	20.7	6.8
Invasive procedure	-	2.6	1.2
Cognitive testing	63.0	51.7	14.7
Questionnaires/surveys	-	60.3	89.6
Remote research	53.6	27.6	46.6
Multiple research visits	63.0	42.2	19.6
Other sample	19.2	-	-

Not all questions were asked to all categories of respondent (noted by -). Corresponding survey questions can be found in the supplementary material under “Survey questions: Previous research participation.” SA, self-advocate; FP, focus person; CG, caregiver.

Fifty-five percent of caregivers reported that their focus person had previously participated in research (Table 2). Caregiver education level (p = 0.082) and age of caregivers (p = 0.108) and their focus person (p = 0.523) were not different between previous research participants and nonparticipants. For focus persons, the most common research procedures included completing questionnaires or surveys, cognitive testing, blood draws, and medical examinations. Sixty-one percent of self-advocates had previously taken part in research (Table 2). A blood draw and cognitive testing were the most commonly reported procedures, and most self-advocates reported participating in more than one research visit.

Attitudes towards research

Tables 3 and 4 show responses to questions asking about attitudes towards research participation for the self-advocate and caregiver groups, respectively. No differences in caregiver or self-advocate response patterns were found based on age or sex. Table S1 shows differences in self-advocate and caregiver response patterns on select questions.

Table 3.

Self-advocate attitudes towards research

“For this part, read each sentence and decide if it makes you want to be in the research study more or less.”
Percentage of total responses	n	Less	No change	More
Practical considerations
Benefits other people with Down syndrome	45	4.4	13.3	82.2
Will receive compensation	45	2.2	46.7	51.1
Takes a long time	45	55.6	42.2	2.2
Welfare of SA
May cause pain	46	45.7	45.7	8.7
Required to do things that are hard	45	62.2	35.6	2.2
CG can stay with SA	46	4.4	23.9	71.7
Research team/procedures
Receive information packet	44	4.6	38.6	56.8
Meet with researcher	45	0	53.3	46.7
Know other participants	46	8.7	43.4	47.8

Corresponding survey questions can be found in the supplementary material under “Survey questions: Attitudes towards research.” SA, self-advocate CG, caregiver.

Table 4.

Caregiver attitudes towards research, shown as a percent of total responses.

“Please rate if the statement would make you more or less likely to encourage the focus person to participate.”
Percentage of total responses	n	Much less	Little less	No difference	Little more	Much more
Practical considerations
Benefits other people with Down syndrome	208	0.48	0.48	2.9	15.4	80.8
Benefits the FP	208	0.96	0.48	7.2	19.2	72.1
Will receive compensation	209	4.8	1.4	47.4	22.5	23.9
Welfare of FP
FP wants to participate	204	0.49	1.5	16.7	31.4	50.0
Research team/procedures
CG interested in topic	209	1.4	1.9	15.3	42.1	39.2
“Look at each statement individually and decide how big of a difference it would make in your feelings about participation.”
Percentage of total responses	n	Much more negative	Little more negative	No difference	Little more positive	Much more positive
Practical considerations
Requires multiple visits	198	20.2	38.4	34.9	4.6	2.0
Welfare of FP
FP does not understand concept	197	9.1	27.4	54.8	4.1	4.6
May be boring or frustrating	199	22.1	57.3	16.1	3.5	1.0
May cause pain	198	61.1	31.3	6.1	0.51	1.0
CG can stay with FP	198	0.51	0.51	6.6	23.7	68.7
FP in other research projects	198	1.5	9.1	55.6	22.7	11.1
Research team/procedures
Receive information packet	196	0.51	3.1	19.9	38.3	38.3
Single research team contact	198	0.51	1.5	17.7	46.5	33.8
CG is included in research	199	0	0.5	16.1	30.7	52.8
Hear from other participants	196	1.0	1.0	40.3	46.9	10.7
Meet with researcher	196	1.0	1.5	30.6	39.3	27.6
Researcher has good reputation	198	0	0	21.2	33.3	45.5
Facility has good reputation	198	0	0	7.1	35.4	57.6
Plan for sharing results	199	0	0	14.6	41.7	43.7

Corresponding survey questions can be found in the supplementary material under “Survey questions: Attitudes towards research.”

CG, caregiver; FP, focus person.

Practical considerations

Caregivers were “much more likely” to encourage the focus person to participate in a research study that had the potential to help individuals with Down syndrome in the future or that would benefit the focus person, such as by providing medical information. Eighty-two percent of self-advocates reported that they would want to participate in a study more if it might help other people with Down syndrome in the future. One caregiver commented:

“Knowing that whatever we are participating in will advance the care, health and lives of other people with DS, or the attitudes of others to people with DS, is what makes us want to participate in a study.”

Both caregivers and self-advocates were split on whether compensation for participation (such as a cash stipend) would make them more likely to participate or would make no difference.

Multiple caregivers cited time and location as critical factors in the decision to participate. Some commenters stated that they would be more receptive to studies that take less time, reflected by the finding that the majority of caregivers would feel negative about participation that required multiple study visits. Others urged researchers to be precise about the time commitment, with one caregiver commenting:

“Some are very open ended, where cognition is tested for 1–2 hours for 2–4 sessions for example. That is a very wide range of a time commitment that is not appealing to me.”

Some caregivers also expressed concerns about the location of the study, stating that extra visits to a medical facility may increase anxiety in the focus person. Citing both convenience and the welfare of the focus person, multiple caregivers stated that coordinating study procedures with clinical care would make them more willing to consider participation, particularly for invasive procedures. One caregiver commented:

“If the more invasive medical procedures needed for research can be combined with doing the same procedure for the individual medical reasons of the ‘focus person’ we would be much more likely to participate.”

Welfare of focus person

Sensitivity to the wishes of the focus person was a critical topic for caregivers. The majority of caregivers would encourage the focus person if they wanted to participate in a research study. In the case of a focus person that cannot make his wishes known explicitly, one caregiver stated that they try to “…respect what […] he might want since he’s largely nonverbal.” The efforts of the caregiver to intuit the wishes of the focus person may partially explain the response to the statement “The focus person has a hard time understanding the concept of research,” which 55% of caregivers said would not make a difference to their feelings about participation.

Comments citing concern for the welfare of the focus person were frequent, including research-related privacy and safety concerns and the risk of exposure to Covid-19 in healthcare facilities. The majority of caregivers reported feeling “a little” more negative towards research that risked causing the focus person boredom/frustration, and the vast majority responded negatively to the risk of physical pain. Self-advocates were comparatively receptive to research procedures that might cause them pain, with 46% less interested in participating and 46% reporting no impact. Doing “things that are hard” for a study was a stronger disincentive for self-advocates. Allowing the caregiver to stay with the focus person during the study was an incentive for the majority of both groups, although more self-advocates reported no impact (Table S1).

Research team and procedures

Respondents were asked about specific actions that may influence the way they feel about participating in a study. Caregivers said that receiving an information packet before enrolling in a study would make them feel more positive about participation. The self-advocate response pattern was similar, with the majority seeing an information packet as an incentive. A single contact person on the research team who would guide the participant through the research process was an incentive to the majority of caregivers, as was including the caregiver in the research (e.g. as an informant).

Learning about the research experience from other study participants was not a strong incentive for caregivers, and a related question about knowing other study participants elicited a similarly mixed response from self-advocates. The opportunity to meet with the head of the study before enrolment elicited mostly neutral to positive responses from both caregivers and self-advocates.

The specific researcher and research topic mattered to caregivers, with the majority feeling more positive about participation if the head researcher and research facility have good reputations in the community. Likewise, a low level of trust in the medical or research community was cited as a barrier by some commenters. Eighty-one percent of caregivers reported feeling more positive about participation when they personally think the research question is interesting.

The positive impact of feeling invested in a research study was apparent in respondent comments, including the drive to contribute to something larger. A plan for sharing the results of a study with the participants was a positive for most caregivers, and many comments underscored the importance of participant communication. One caregiver commented:

“The last large research study we participated in suddenly closed and we were not notified. It felt like all of our time and effort were meaningless and we were not even important enough to inform of the closing much less the reason why it was closed.”

This comment also touches on the theme of respectful treatment. One respondent noted that they are sensitive to how the research team interacts with the focus person, and another suggested that they would like the research team to show “positive and grateful attitude[s]” towards the participants. Several commenters pointed out that people who are nonverbal or have a dual diagnosis (e.g. Down syndrome and autism) have a barrier to participation imposed on them, as many studies will exclude these participants.

Specific procedures

Caregivers were presented with specific research procedures and asked how likely they would be to allow the focus person to undergo the procedure for research purposes (Fig. 2). The most readily accepted procedure was cognitive testing, followed by medical examinations, collection of urine samples, psychiatric interviews, and blood draws. Caregivers were mixed on medical imaging and unspecified procedures requiring local anaesthetic, and most caregivers would be unlikely to allow lumbar punctures or unspecified procedures requiring full sedation.

Figure 2.

Caregiver attitudes towards specific research procedures. Caregiver response to “Please rate how likely you would be to allow the focus person to enroll in the research and undergo the procedure.”

Information sources

Finally, we asked caregivers how they receive and share information about Down syndrome (Fig. 3). The most common sources were online groups related to Down syndrome, newsletters or outreach from local or national advocacy organizations, other caregivers, and social media. Fewer caregivers reported receiving information from their local government agency serving individuals with intellectual or developmental disabilities or from the development of an individualized plan for the focus person.

Figure 3.

Caregiver responses regarding how often they use specific sources to share and receive information related to Down syndrome. ID, intellectual disability; DD, developmental disability.

Discussion

Researchers have a responsibility to consider the opinions of all major stakeholders, including individuals with Down syndrome, their families, and their caregivers. This report adds to recent surveys of parents and carers by facilitating individuals with Down syndrome to contribute their views to the discussion of motivators and barriers to research participation.

In this study, most caregivers reported that they had previously participated in research, with approximately half of these previous studies consisting of surveys. This number was likely affected by the use of DS-Connect for recruitment, as DS-Connect registrants are offered online surveys as a part of the registry. Self-advocates also reported a relatively high rate of previous participation, and the rate for focus persons was higher than the 36% previously reported by White et al. (2022). It is possible that respondents who are interested in or have experience with research were more likely to complete this survey, although these results are consistent with the high level of research interest reported among carers of children with neurodevelopmental disorders (Kalb et al., 2019).

The potential to improve the quality of life for other people has been repeatedly cited as a motivator to research participation (Haas et al., 2016; McDonald et al., 2013; White et al., 2022). Both this altruistic aspect and the social aspect of feeling like a part of a purposeful community emerged as strong themes in this analysis, as did the importance of trust, which has been highlighted in previous studies of research intellectual and developmental disabilities (Haas et al., 2016; McDonald et al., 2013). Caregivers expressed a desire to feel invested in research, whether through understanding the purpose of a study and learning about the results or receiving regular communication and feeling like a part of a research community. Communication and community building can take many forms, including regular newsletters, hearing from other participants, or holding a dedicated meeting to discuss the findings of the research (Robinson et al., 2007). These efforts could take advantage of commonly used information sources such as online groups related to Down syndrome, local and national advocacy organizations, and social media.

Sensitivity to the wishes of the focus person was paramount for caregivers. Consistent with White et al. (2022), we found that risks to the participant strongly affected the decision to participate. Most caregivers responded negatively to procedures generally perceived to be more painful or risky, such as lumbar punctures (Kyprianou et al., 2023; White et al., 2022), while self-advocates were more neutral towards the risk of pain. Similarly, Kyprianou et al. (2023) reported that some caregivers did not ask the focus person to participate in research because they believed they would not be interested in participating. Caregivers understandably seek to shield their focus person from discomfort or distress, but these findings suggest that an open discussion of research procedures, including exactly what each entails and possible risks, is the best way to empower participants to state their opinions and come to a mutual decision with their caregiver. Researchers can facilitate these discussions by providing thorough yet easily understandable descriptions of each procedure, taking advantage of pictures or props to facilitate understanding. Providing clear descriptions can also address research hesitancy in caregivers, targeting common misconceptions or concerns about specific procedures.

For caregivers, altruistic motivations were balanced by practical considerations. Consistent with other investigations, we found that barriers to participation include time and travel considerations (Haas et al., 2016; Quinn & Menon, 2020; White et al., 2022). Interestingly, White et al. (2022) reported that parents were more positive towards studies with a longer duration, such as longitudinal studies lasting one year or longer, whereas this survey found that multiple study visits were considered a barrier to participation. It is possible that the wording of the questions contributed to this difference. For example, the phrase “multiple visits” may have been more readily associated with a greater time commitment than the phrase “longitudinal participation.” Spanning considerations both practical and related to the welfare of the focus person, multiple commenters suggested that merging clinical care with research would facilitate and incentivize participation, particularly if research and clinical visits were combined and clinically required procedures leveraged for research purposes.

Through survey questions and comments, respondents indicated how investigators could improve the research process. Receiving an information packet about the study was a popular idea, with commenters suggesting the inclusion of practical information such as a study timetable, maps, or information about the local area for those who are travelling. Caregivers also responded positively to the idea of having a single contact person to guide them through the research process, consistent with the personal approach described by Nicholson et al. (Nicholson et al., 2013).

A limitation of this work is the restricted generalizability of the responses. Survey respondents were predominately Caucasian, with most caregiver respondents being mothers with a college degree or higher. Our sample was likely biased towards those with an interest in research; attitudes towards research may differ in the general population. Efforts to broaden respondent demographics and engage those not familiar with research are required to fully assess the needs and desires of a diverse population. Such efforts could include setting up response booths at community functions related to Down syndrome, partnering with clinicians to collect responses at appointments, making the survey itself more inclusive (for example, by making a Spanish language version available), and engaging self-advocates as co-researchers (O’Brien et al., 2014).

Conclusion

Although the decision to participate in research is complex, this survey finds that both caregivers and adults with Down syndrome are strongly motivated by the personal and community benefits of research. Caregivers also indicated the critical need to be sensitive to the wishes of the person that they care for. Our results hint at a possible difference in risk tolerance between adults with Down syndrome and their caregivers, highlighting the responsibility of researchers to engage all parties in open, clear discussions of research procedures.

Data Availability Statement

The data used in this analysis is available via request of the corresponding author.