Engaging youth and parents in clinical pediatric research: A case-based example

Abstract

Background

Youth and parent engagement is a key component of clinical research. There are many ways to actively and meaningfully engage youth and parents as integral members of research teams, for example, through ad-hoc committees, advisory councils, or as co-leads on projects. When youth and parents are actively and meaningfully engaged in research projects, they share knowledge from their lived experiences to improve the quality and relevance of research.

Methods

We describe a case-based example of engaging youth and parent research partners when co-designing a questionnaire to assess preferences for pediatric headache treatments, from both a researcher and youth/parent perspective. We also summarize best practices in patient and family engagement from the literature and pertinent guidelines to assist researchers with integrating patient and family engagement into their studies.

Results

As researchers, we felt that the integration of a youth and parent engagement plan into our study significantly altered and strengthened questionnaire content validity. We encountered challenges throughout the process and detailed these experiences to help educate others about challenge mitigation and best practices in youth and parent engagement. As youth and parent partners, we felt that engaging in the process of questionnaire development was an exciting and empowering opportunity, and that our feedback was valued and integrated.

Conclusions

By sharing our experience, we hope to catalyze thought and discussion around the importance of youth and parent engagement in pediatric research, with the goal of stimulating more appropriate, relevant, and high-quality pediatric research and clinical care in the future.

INTRODUCTION

Engagement with youth and parents across the lifespan of research projects is imperative to ensure their voices and perspectives inform research processes. Youth and parent engagement in research has also been shown to lead to better quality research by improving recruitment rates, and by producing meaningful, stakeholder-led change in research protocols and outcomes (1–3). Two ways to engage youth and parents are (1) as participants, described as those who enroll in research and play a more passive role, or (2) as partners for which level of engagement is on a spectrum (4) from minimal involvement as research participants toward higher levels of engagement on ad-hoc committees, standing advisory councils, or co-led projects (5,6). Higher levels of engagement occur when there is meaningful and active participation during priority setting, while preparing to conduct research studies, when collecting data or assisting with analysis, or while planning knowledge translation activities (7–11). Careful planning is needed to ensure that the level of engagement is suited to youth research partners and that it remains authentic (3). For example, involving youth in the decision-making around the type of role they will undertake, and how active they would like to be, is helpful to achieve optimal engagement; more active roles generally lead to better engagement outcomes (3). Initiating engagement can be done via consultation with youth advisory boards; however, other recruitment means can be used including advertisements, recruitment through community groups, or through patient advocacy networks (9). The findings from a scoping review showed that youth and parents felt more empowered to make changes to their own health when they saw changes in the research process as a result of their engagement (11).

Here, we describe a case-based example of youth, parents, and researchers co-designing a patient preferences questionnaire to evaluate treatment options for primary headache disorders. The aim of the questionnaire is to assess initial treatment and outcome preferences of youth with primary headache disorders and their parents in the clinical setting, so as to guide patient- and family-centred decision-making. We also share what we learned through the engagement experience as researchers, youth research partners, and parent research partners on this project. We describe the benefits of the engagement experience during the co-development of this questionnaire, challenges that arose, and propose some actionable items from our experience that are aimed at helping researchers engage youth and parents effectively in their studies. Equally important were the values that guided our engagement experience in this project, which were an openness to learning from each other’s experiences and flexibility with the process of engagement.

METHODS

Sampling, recruitment, and participants

As researchers, we initially developed our youth and parent engagement plan with the assistance of knowledge brokers within the Alberta Strategy for Patient Oriented Research Support Unit (AbSPORU). We aimed to recruit 5 to 10 youth and parent advisors. There is no optimal group size for patient and family engagement, and most engagement studies emphasize the need for a safe space that encourages discussion from both researchers and partners (9), which may be more attainable in smaller groups. For the purposes of our project, we identified “youth” as any person under 18 years of age. “Parent” described any person, caregiver, or legal guardian in a primary caregiving role for youth. Parents and youth involved in the questionnaire development were not related.

For youth partner recruitment, we consulted with KidsCan Young Person’s Research Advisory Group (YPRAG), a pediatric research advisory group comprised of Canadian youth 13 to 19 years of age. For parent partner recruitment, we connected with the Pediatric Parents’ Advisory Group (P-PAG), a group of parents, legal guardians, and grandparents who serve as advisors to pediatric research programs at the University of Alberta. We did not specifically target the recruitment of parents of youth with headache disorders, but some of the recruited parents did have this lived experience. Unfortunately, our original plan to recruit parents from P-PAG did not yield sufficient parent partners (N = 2). This was likely in part due to competing priorities during the COVID-19 pandemic (parent sessions were held in the spring of 2020). In order to recruit a sufficient number of parent partners to capture diverse perspectives, we then used social media (Twitter) and targeted parents in the researchers’ local community and work settings.

Informed consent process

Written informed consent was provided by all youth and parent partners for participation in this project. This study was approved by the Conjoint Health Research Ethics Board (REB19-1319).

Study design

As a first step in the development phase and to facilitate discussion during youth and parent engagement, the research team drafted initial questionnaire items based on information required to appropriately inform and counsel patients in the clinic. Next, we solicited feedback from 18 pediatric headache specialists across North America via email, which was incorporated into the subsequent draft. As researchers, we chose to consult with the expert group first given significant growth in headache treatment options over the last 5 years; in this context, we felt that expert consultation was the most appropriate first step.

Prior to our engagement sessions, we sent the draft questionnaire to YPRAG and parent partners via email. We held two 90-minute videoconferences with each advisory group to explain the aims of our project and discussed the draft item by item. For those unable to attend, we hosted separate video- or teleconferences. We had group discussions regarding questionnaire appropriateness, content, clarity, feasibility, the language, and goal of each question. For those unable to attend the videoconference, we sent a brief survey with the following two questions:

1. Please provide feedback about the items contained in Sections A and B of the “Patient Preference Questionnaire”. Are there any items that could be improved for clarity, wording, or validity?

2. Please indicate if you feel that any items should be added or removed to improve the “Patient Preference Questionnaire”.

Researchers then invited youth and parent partners from these engagement groups to co-author this manuscript, detailing our experience in co-designing the questionnaire. Eight of the 22 youth and parent partners expressed interest. The initial manuscript was drafted with the researchers generating initial content for all sections except for the youth and parent perspectives, generated by each group, respectively. All co-authors then reviewed, edited, and commented on the entirety of the manuscript until all felt that each perspective was adequately reflected throughout.

Analysis plan

Once all feedback had been obtained from youth and parent partners, we organized the feedback into similar themes. Where distinct themes arose from youth or parent partners, the researchers met to discuss how to resolve the conflicts.

Results

Development of a youth and parent engagement plan

We partnered with experts in patient engagement from the AbSPORU, as described above, to develop our youth and parent engagement strategies and goals. This approach was aligned with our values of engagement in that we were open to learn from experts in engagement and were flexible with the engagement plan. Through this partnership, we were encouraged to evaluate both youth and parents’ perspectives to represent both individual patients’ and families’ expectations. These groups’ perspectives allowed for better alignment of researchers’ priorities and goals to those of the target population in headache clinics (12).

The recruitment of youth and parent partners:

We recruited YPRAG members comprised of six youth and five YPRAG coordinators from across the country (N = 11). Two of the youth had lived experience with headache disorders. We recruited 12 parent partners, 2 of whom had children with lived experience of headache disorders, and 3 of whom were healthcare providers (N = 12). Though the proportion of youth and parent partners with lived experience was proportional to the population prevalence of headache disorders (13), we could have had a better representation of individuals with lived experience in this partnership. In future endeavours, we plan to use a more targeted recruitment strategy to specifically identify partners with lived experience.

As a research team new to the youth and parent engagement process, recruitment was challenging. This is not a unique phenomenon; prior summative evidence suggests that recruitment of youth partners is often a significant challenge in pediatric studies, but that working with existing patient engagement groups, as we did, can facilitate the process (9). The ambiguity about how to proceed with consent also resulted in modifications to ethical application processes. Specifically, we were initially unaware that consent would be required for the planned youth and parent engagement. Once this was clarified by our local research ethics board, we sought consent retrospectively. In the future, we will proactively discuss consent requirements prior to initiating engagement. Another challenge pertained to deciding on how best to connect with partners. In the end, we used teleconference, videoconference, and email surveys to engage with national partners, which required a substantial amount of time to coordinate but maximized options for our partners. These experiences demonstrated that we were open and flexible to making changes to our engagement strategies so that we could enhance the experience with everyone involved.

Although we had a group of youth partners who were diverse in gender (n = 8 female; n = 3 male), ethnicity (n = 8 Caucasian, n = 3 Other), and geographic location (pan-Canadian), we did not collect demographic data from parent partners, which limited reporting and assessment of the parent group’s composition. A scoping review on this topic highlights strategies for ensuring that diverse youth are recruited, including diverse participation opportunities, and involvement of community organizations in the recruitment plans (9). We plan to use these strategies in the future.

Implementation of youth and parent engagement:

A summary of the feedback provided and how it was used to improve the questionnaire is available in Table 1. During initial questionnaire development, we sought input from YPRAG on establishing an appropriate lower age range cutoff for inclusion in our study, and the group (aged 13 to 19 years old) felt that our target age range of 10 to 18 years was appropriate after their input. As an example, the youth indicated that using percentages to depict headache frequency was appropriate for the target age range and provided guidance on how to present numerical items to younger participants. Youth with lived experience emphasized the importance of having the impact on school performance included as an option for outcomes to assess.

Table 1.

Examples of the engagement group’s contributions.

Engagement Group Feedback	Changes Made
Some treatment options were difficult to understand.	A legend was included at the beginning for reference and more descriptors were added to questions.
Youth felt they preferred ranking items related to treatment preferences in order of preference rather than rating each item on a Likert scale.	Similar questions formatted as Likert scales were eliminated, and replaced with questions that allow participants to rank treatment preferences.
	Changed sentence wording for this item asking for the longest amount of time acceptable.
Choosing “top three” side effects to avoid was difficult.	Wording was changed to “choose three”.
Discussed percentages versus fractions and youth reported percentages were easier to understand.	Removed alternative question.
Choosing from the treatment option list could be overwhelming.	Added a “I don’t know” option with free text to explain.
Parent groups felt that quantifying risk of side effects such as “low versus high risk” was difficult to understand.	Removed risk quantifiers in the questionnaire items discussing side effects, leaving this open to further discussion in clinic.
Parent groups had difficulty thinking about potential side effects.	Included a disclaimer indicating that all treatments had side effects.

Two themes that arose from the parents’ feedback on the questionnaire were (1) difficulty interpreting potential side effects of treatment and (2) difficulty wading through the number of side effects of treatment. Both youth and parents suggested that more context and descriptors about treatment options and side effects were needed. This feedback led to adjustments in the wording and to the creation of a legend at the beginning (e.g., description of what an injection is, what acute versus preventive treatment is, etc.). Parent partners with children with headache disorders also provided unique perspectives indicating the comprehensiveness of the described treatment options was valuable for parents preparing for a consultation with a neurologist. Overall, when asked if the groups thought the questionnaire addressed the aims of our study, the sentiment was unanimous and all questions were felt to be necessary and appropriate. This outcome was possible in part because of our values of engagement, in that we were open to hearing and incorporating the perspectives from parent partners and children, so that the questionnaire would reflect what was important to them.

We encountered challenges during the implementation phase. One example was the tension between addressing single partner’s comments or relying on common themes emerging. For example, a youth suggested adding a question to reflect parent income since they wondered if finances would influence treatment preferences. This was discussed with the parent group and they felt this question would create unnecessary bias in how parents respond to the questionnaire. Ultimately, given the strong parental feedback, we chose to not add an item on parental finances. Some of the parents had previously participated in similar research engagement opportunities and could therefore provide recommendations based on past experiences (e.g., around appropriateness and feasibility of number of questions and time required for questionnaire completion). Some youths had experience with completing questionnaires in other medical clinics and provided informed feedback based on this experience.

Discussion

Perspectives on our partnerships

From the researcher perspective.

Family-centred research helps to align researchers’ and families’ priorities (14) and, in our case, provided youth and parents with an opportunity to engage in research aimed at better understanding their lived experience. These partnerships were instrumental in ensuring that our questionnaire addressed questions that were not only important to clinicians but also to youth and parents. The engagement experience allowed for important modifications that were not intuitive from the researcher’s lens. Managing the back-and-forth discussions between and within groups was challenging but yielded richer results and better inclusion of all parties involved. We strove to acknowledge, welcome, and respect the lived experience that youth and parents provide as this approach has been shown to result in more shared decisions and to decrease power imbalances (15). Overall, we felt that the inclusion of both youth and parent partners in our questionnaire development significantly strengthened the content validity of our questionnaire and was valuable in capturing a variety of perspectives.

From the youth perspective.

As some of the youth partners have experience with chronic headaches, we arrived at the consultation with personal experience. Therefore, the experience of being involved in this research is very meaningful. It is exciting to see what researchers are doing in this field to come up with new strategies for treatment and management involving patients as partners. Being involved in the research also provides us with a way to see change happening in the field. Through observation of active change making, it also fuels advocacy efforts on the grassroots level. As youths with lived experience, it was encouraging to see that there was progress happening in this area (i.e., headache medicine). As youth grow up, it is important for them to advocate for their own health care and being involved in research demonstrates to youth that their perspectives matter and are valued. Our group at KidsCan felt that the consultation on this study went smoothly, with the researchers presenting adequate pre-consultation materials in a way we could understand and readily answer questions on the day of the consultation. It was helpful to be part of preparing the manuscript and we look forward to the researchers providing us with a list of direct impacts from our contributions when the project is finished.

From the parent perspective.

From the parent research partners’ lens, co-designing the questionnaires was a rewarding process. As some of us are parents of youth with chronic conditions, we provided perspectives from our real-life experiences, different than those of healthcare providers. However, it is important to note that some of us are also healthcare providers with shared lived experiences of caring for someone with a disability or living with one ourselves and we were thus able to provide unique perspectives. Having parents and other caregivers co-design this questionnaire allows healthcare providers to gain further insight into the appropriateness of their evidence-based treatments and allows for a more comprehensive and useful tool to be prepared from the start. The structure and functioning of the development team also reduced the power imbalance that can exist between healthcare providers and parents/caregivers by regularly implementing parental feedback into the survey. This experience also allowed the parents to provide significant input around how they may weigh treatment goals against risks/side effects of treatment based on their perspectives and exposure to the questionnaire. Incorporating these parental perspectives and risk/benefit analyses into the discussions helped us to feel that the power imbalance could be reduced in clinical encounters by increasing parental engagement in decision-making.

SUGGESTIONS FOR FUTURE STUDIES

Some key lessons learned from our experience include the importance of:

1. Taking adequate time to develop an engagement plan, including discussion regarding the planned level of engagement and roles prior to starting engagement;

2. Consulting with experts in knowledge translation and patient engagement as needed, particularly when the research team is new to patient and family engagement;

3. Identifying a diverse engagement group to ensure diverse perspectives;

4. Deciding on ways to navigate opposing ideas and fair integration of everyone’s ideas; and

5. Including a way to evaluate the youth and/or parent engagement process as this is a large gap in the current literature (9).

We also hope to summarize some tips about how to engage youth and parents in pediatric research based on best practices from existing guidelines, which are in Table 2. We recommend the Canadian Institutes of Health Research’s Strategy for Patient-Oriented Research (CIHR-SPOR) as a key reference for individuals wanting to learn more about patient engagement in research (https://cihr-irsc.gc.ca/e/48413.html).

Table 2.

Suggested best practices for family engagement in future projects.

1. Familiarize yourself with patient engagement frameworks. As examples, the CIHR has a Strategy for Patient-Oriented Research (SPOR) available at https://cihr-irsc.gc.ca/e/48413.html, and PCORI has information and resources at: https://www.pcori.org/engagement.

2. Think about youth and parent engagement as early as possible during the planning phase and protocol development and budget for resources (in-kind and monetary).

3. Contact your local university-affiliated pediatric research group to inquire about local expertise in youth and parent engagement. If you are carrying out research in Canada, connect with your provincial Strategy for Patient-Oriented Research (SPOR) Support unit contact, which is found on the CIHR website (https://cihr-irsc.gc.ca/e/45859.html) or PCORI (https://www.pcori.org/) if you are in the USA.

4. Decide whether to use existing youth or parent groups or to create a new panel. If you aim to create a new panel, search for knowledge mobilization networks in your area of research who may assist you with engagement and panel recruitment while incorporating an equity, diversity, and inclusion lens (e.g., SKIP: https://www.kidsinpain.ca/, TREKK: https://trekk.ca/researchers).

5. Strive to integrate youth and parent engagement in as many steps of your research as possible, while keeping feasibility in mind (see the How-to-Patient Engagement Guide for Patient Partners and Researchers by AbSPORU).

6. Include a way to evaluate the impact of youth and parent engagement in your research (review the extensive toolkit here https://ceppp.ca/en/collaborations/evaluation-toolkit/ for one that would fit your research study) (16).

7. Include the eventual partnership model in your protocol submission to ethics. Youth and parent partners can be listed as co-investigators, collaborators, extended research team members, or as an advisory group.

8. Invite youth and parent partner(s) from your projects to promote your shared experience with other research teams and the public through podcasts, scientific and public presentations, blogs, social media narratives, and scientific articles.

9. Include youth and parent partners in planning and deciding on appropriate knowledge translation activities, which will help with sharing and implementing the findings from your studies (e.g., develop an infographic, create an animated video, share key tips on a site for youth).

AbSPORU, Alberta Strategy for Patient Oriented Research Support Unit; CIHR, Canadian Institutes of Health Research; PCORI, Patient-Centred Outcomes Research Institute; SKIP, Solutions for Kids in Pain; SPOR, Strategy for Patient-Oriented Research; TREKK, Translating Emergency Knowledge for Kids.

CONCLUSION

In clinical pediatric research, youth and parents are key knowledge users, and researchers should strive to engage with them as much as possible in the various phases of research design, implementation, and knowledge dissemination in order to optimize the relevance, feasibility, and impact of their research (1–3). By sharing a case-based example rooted in our recent experience with youth and parent engagement, we hope to have highlighted the importance of such partnerships, the benefits of a multimodal approach to engagement, values of engagement to espouse toward, and lessons learned from the challenges that we faced as researchers who are relatively new to youth and parent engagement. The more we can encourage clinical researchers at any stage of their careers to pursue youth and parent engagement, the more we can foster a future with more impactful research. Going forward, we hope to hear more discussion around researchers’ experiences with youth and parent engagement, to see more research into optimal engagement strategies, and to see better education of researchers regarding how to best engage with youth and parents.

Abbreviations

AbSPORU

Alberta Strategy for Patient Oriented Research Support Unit

CIHR

Canadian Institutes of Health Research

PCORI

Patient-Centred Outcomes Research Institute

P-PAG

Pediatric Parents’ Advisory Group

SKIP

Solutions for Kids in Pain

SPOR

Strategy for Patient-Oriented Research

TREKK

Translating Emergency Knowledge for Kids

YPRAG

KidsCan Young Person’s Research Advisory Group