Introduction

Patient-oriented research focuses on engaging patients in the research process. This type of engagement helps to ensure that research answers questions relevant to patients and uses collaboratively developed methods, and interpretation of results, with the goal of improved patient outcomes [1]. Though efforts to conduct patient-oriented clinical trials have increased [2], the degree of engagement in patient-oriented research is paradoxically poor [3] or may appear tokenistic. Patient engagement is intended to align research goals between researchers and patients, ensure research design and methods are feasible and accessible, and facilitate effective dissemination of study findings to those who contributed data as well as other stakeholders and the public. Patient engagement research has the potential to be especially impactful in trials focusing on young children, where parents can develop ongoing and genuine interactions with study teams, including clinicians and researchers, and provide meaningful input such as steering study design and dissemination of results. There is limited literature on engaging parents and young children as partners in research involving young children [4,5,6,7].

The Applied Research Group for Kids (TARGet Kids!; www.targetkids.ca) is an ongoing cohort study with embedded randomized controlled trials (RCTs), which involves parents and young children from primary care practices in three cities in Canada (Toronto, Montreal, and Kingston). TARGet kids! is a collaboration between parents, their children, child health researchers, and primary care practitioners, which aims to link early life exposures to health problems including obesity, micronutrient deficiencies, and developmental problems [8]. TARGet Kids! presents a unique opportunity to engage parents and inform research priorities, improve study recruitment and generalizability, and co-develop embedded clinical trials. One embedded randomized controlled clinical trial in TARGet Kids! is called PARENT (Parenting Addressing Early Years Intervention with Home Visits in Toronto: A Pragmatic Randomized Controlled Trial; NCT03219697), a pragmatic, parallel-group, one-to-one, superiority RCT. The aim of the PARENT trial is to determine if participation (experimental group) with a public health nurse via group parenting sessions (i.e. coaching calls addressing goal setting), and home virtual visits (i.e. addressing behaviours in the home setting) focused on nutrition, physical activity, and sleep among young children at risk of obesity can improve health outcomes such as reduced risk for overweight or obesity, and better mental health and nutrition. The control group comprised of children and parents who received usual care. In Canada, this consists of individual well-child health supervision visits, guided by the Rourke Baby Record (as endorsed by the Canadian Pediatric Society and the College of Family Physicians of Canada) [9]. Ethical approval for the PARENT trial and supporting material was received from the Hospital of Sick Children’s and the Toronto Public Health (Toronto, CANADA; REB1000054998). Informed consent was obtained from the parents of children who participated in this study.

In accordance with the International Association of Public Engagement (IAP2) [10], researchers in TARGet Kids! aim to inform, consult, involve, collaborate, and empower parents as co-creators. Thus, the PARENT trial was designed with the support of and in collaboration with parents of young children. Models of co-designing clinical trials are increasingly being seen as a way of addressing power imbalances by designing and delivering research in more democratic, equal, and reciprocal relationships between healthcare professionals, researchers, participants, and end-users [11]. While there have been speculations on the “best” or “correct” way of engaging in co-production and co-development and assessment of clinical trials [5], some key principles that help underpin this practice include developing peer support and engaging a range of stakeholder networks inside and outside health care; removing tightly defined boundaries among researchers, practitioners, and recipients such as parents; enabling shared control, responsibility, and mutuality; and supporting reciprocal relationships with mutual responsibilities and expectations [12]. It has been well documented that the priorities of researchers may not align well with the priorities of patients and clinicians [13,14,15,16]. Patient involvement in setting research priorities may lead to research that is more relevant to patients [17, 18]. Although patients and clinicians are increasingly recognized as important research partners, a review of priority setting studies found that only 49 of 258 (19%) studies involved both patients and clinicians in the process [19]. Nevertheless, evidence generated from partnering with parents to design and conduct research together may be used to refine, adjust, and modify future research approaches, and provide an opportunity to evaluate the process taken to engage patient partners and determine areas of strength, weakness, and need for growth.

The primary objective of this study was to describe the initial approaches to parent engagement in the design of the PARENT trial in TARGet Kids!. The secondary objectives were to understand parent perspectives on the acceptability and relevance of the PARENT trial and potential barriers and facilitators to participation, though a parent focus group.

Methods

Approach to patient engagement in co-design of the PARENT trial

A priority setting process was conducted to identify parent and clinician research priorities in prevention research. Using the James Lind Alliance [20] framework, this exercise revealed that the prevention of obesity and mental illness in young children were two high priority areas worthy of further investigation [21]. This was consistent with priorities of the WHO [22], Public Health Agency of Canada [23], Public Health Ontario [24], and the Canadian Task Force on Preventive Health Care [25], as rates of overweight and obesity in children under 5 years of age in Canada are currently 15% and 6%, respectively [26, 27].

Pilot work was initiated to develop an RCT to promote health behaviours and prevent obesity in young children with increased risk factors for future obesity. Existing literature was reviewed and identified using home visits was effective in trials that addressed parenting strategies for obesity prevention [28,29,30,31,32,33]. Funding for the PARENT trial was obtained through the Ontario Child Health SUPPORT Unit (OCHSU) Strategy for Patient-Oriented Research (SPOR) research network, which encouraged partnerships with patients and other patient-oriented research groups (clinicians and researchers with expertise in public health, primary care, nutrition, physical activity, mental health, early child development, obesity treatment, and epidemiology).

PARENT panel in the TARGet Kids! cohort

Parents participating in the TARGet Kids! cohort were invited to participate in a focus group, called the PARENT panel, to co-design the PARENT trial. This focus group intended to capture diverse individual and collective parents’ experiences. Overall methodological approaches for the PARENT panel were informed by the CIHR Strategy for Patient Oriented Research (SPOR) guiding principles (mutual respect, co-building, inclusiveness, and support) [34] for patient engagement in research, and facilitated through the Knowledge Translation Program in the Li Ka Shing Knowledge Institute at Unity Health Toronto [34]. Along with the PARENT panel, the research team designed an engagement plan inspired by the IAP2 framework to help direct this work [35]. Using a Nominal Group Technique [36], the PARENT panel helped provide feedback on the feasibility, relevance, and acceptability of the proposed intervention. The PARENT panel prioritized research outcomes in the PARENT trial and contributed to the list of potential child and family barriers and facilitators to participating in the intervention. Through informing how to tailor both TARGet Kids! and the PARENT trial to increase participation, parents had the opportunity to co-build the PARENT trial research to meet their interests and needs.

Participant recruitment for the PARENT panel

Recruitment advertisements were posted in TARGet Kids! primary care practices and on the TARGet Kids! website as well as sent via email to TARGet Kids! participants. An online survey was used to determine eligibility (must be involved in TARGet Kids!, have a child between age 12 months and 6 years, have a computer and internet access) and gain informed consent. Background information about the PARENT trial was shared via email to interested and eligible participants prior to the focus group to enable them to contribute to the discussion. Participants received a $15 gift card for local grocery store for participating.

Data collection and analysis

Once informed consent was obtained, a 60-min focus group was conducted using WebEx, an online web conference platform, which enabled participants to review a brief video presentation about the PARENT trial prior to the focus group discussion. PARENT panel participants were engaged in meaningful discussions through open-ended questions facilitated by a trained qualitative moderator, using a 10-item semi-structured interview guide created by the Knowledge Translation Program (in consultation with the PARENT research group; Appendix). The focus group discussion was digitally recorded and transcribed verbatim by a member of the Knowledge Translation Program and member-checked by participants prior to analysis.

Data analysis was informed by the Theoretical Domains Framework (TDF; average silhouette value 0.29, good face validity) [37] to determine root causes of barriers and facilitators to engagement in the PARENT trial. The TDF involves coding interview transcripts into theoretical domains (deductive analysis) and is well-suited for explaining implementation problems and informing implementation interventions [37]. Data were analysed by generating overarching themes from the selected theoretical domains (thematic inductive analysis). The overarching themes represent the factors which were perceived to influence performance of the target behaviour. Once identified, themes were reviewed by the study team to develop and apply changes to the PARENT trial protocol to ensure parent views and suggestions were incorporated. The SPOR Patient Engagement Framework [34] was also used to align and support the synthesis of participants’ recommendations for increasing engagement in the PARENT trial.

Results

In 2019, a total of seven parents (mean age = 31.4 [6.2] years, 100% female, 71% European descent, 86% employed full-time) were recruited for participation in the focus group. As evidenced by supporting quotations regarding trial perceptions, the PARENT Trial was well-received by participants (Table 1). Overarching themes related to increasing parent engagement in the PARENT trial (i.e. barriers and facilitators) emerged from the focus group transcript (Table 2) and are described below according to different aspects of the study.

Table 1 Parent perceptions of the PARENT trial
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Table 2 Elicited barriers and facilitators from participants regarding the PARENT trial
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Target condition

Participants felt the trial would be effective in attenuating childhood overweight and obesity. They believed the material provided was detailed, evidence-informed, and actionable (i.e. provided many useful tips). Home visits were highlighted as being especially beneficial as they encourage accountability and offer assistance. Participants felt that visits from public health nurse would provide a unique opportunity to ask questions, receive support, and add accountability to the proposed behavioural changes. The primary reservation noted by participants was the use of BMI to assess weight-related outcomes in children. Participants questioned its utility in the study and were less convinced that it was the most appropriate measure to evaluate outcomes of overweight and obesity in children.

Foster opportunities for peer support (TDF domain: “social or professional role and identity” and “emotion”)

Parents were interested in engaging with and learning from other parents who were going through similar experiences with their children (e.g. developing an online platform to share experiences). They believed that the PARENT trial had great potential to enhance group connectedness if given the opportunity to interact with other participants’ parents: “...it’d be interesting to have that social engagement with the parents as well, and have parents interact with each other (maybe swap tips and stuff like that). I don’t know if that’s possible, but it would make it more interesting” [P2].

Memory, attention, and decision processes

Participants felt it may be difficult for parents to differentiate between the different trials or studies being offered through their primary care physician’s office and at times felt overloaded with the amount of similar information being shared. However, participants also noted that because the request to participate was coming from their healthcare clinic (wherein they already a trusted and established relationship), they may be more likely to partake in the trial.

Trial design

Focus group respondents provided detailed recommendations for improving the recruitment, engagement, and intervention of participants in the PARENT trial (Table 3).

Table 3 Parent-reported PARENT trial recommendations
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Use alternate communication methods for participant recruitment (to distinguish between different trials within the TARGet Kids! cohort; TDF domain: “environmental context and resources”)

Parents reported that trial recruitment materials were not easy to differentiate from other emails related to the ongoing cohort study. Suggestions for methods that may be more effective in gaining attention, as stated by one participant: “... email can sometimes get lost in my inbox, so I would prefer something like a hard mail copy or something directly in the Physician’s Office...” [P5].

Share detailed information about potentially invasive trial activities (TDF domain: “Beliefs about the consequences”, “knowledge”, and “reinforcement”

Parents recommended sharing detailed information about the purpose of, and protocol for, potentially invasive components of the trial. For example, many parents had concerns regarding their young children undergoing blood tests related to obesity outcomes, as expressed by “The things that I don’t like [laughing] just being honest, I’m a little bit nervous about the blood sample that’ll be taken, but that’s as a first-time mom – just scared about that for my little one” [P3] and “…in the control group it just feels a little bit extra like “ugh, so we got to give the blood but we don’t get the at-home visits or the one-on-one support” so I guess that’s what felt like a potential harm for me” [P5]. Home visits by public health nurses were also noted as a friction point: “In terms of not like: I can see people being concerned about the home visits – about “what are they looking for? Are they going to judge the state of my home?, you know, just things like that that people I know can be concerned about when people are coming into their homes” [P1] and “I would also have concerns if there were home visits – I think my worry would be ugh, what are they looking at? Are they going to be looking at my sink dishes in it, or are they judging the whole atmosphere here, or are they just sitting to have the talk? So hearing what to expect” [P3].

Consider professional role in the discussion of sensitive information (TDF domain: “social or professional role and identity” and “memory, attention, and decisions processes”)

Parents identified that as a participant in the PARENT trial, they would want to receive information about why they were being recruited to participate and to be able to discuss any related medical concerns with their physician. One parent noted “I think the only thing that’s stood out for me is: reading in the protocol (or the consent) that it’s specifically for kids who are at a high-risk for childhood obesity, and I think when I read that I was like “what? My kid’s at high-risk for obesity? I didn’t even know that” so just some questions that come up with recruitment around that” [P4].

Intervention

Belief about consequences

Panel participants were apprehensive about having their young children undergo unnecessary blood work. They were nervous about the undue pain and discomfort this might cause for their children.

Emotion

Due to the young age of the children, parents expressed negative feelings about their kin providing a blood sample and elicited negative emotions, including nerves and fear.

Reinforcement

Parent panellists expressed that those assigned to the control group may be less likely to have their child provide a blood sample as they will not be receiving anticipated trial benefits allotted to those in the experimental group (e.g. home visits, support, group sessions, information) and thus reinforce their perception of harm.

Social or professional role and identity

‘Who’ was delivering the intervention was raised as an important point. While some felt that the approach of public health nurses may not be congruent with the nature of support parents want to receive in the home visits (and that a social worker or nutritionist may be better), whereas others perceived the expertise and approach of public health nurses to be appropriate for their needs.

Knowledge

Panel participants noted a lack of understanding regarding what the public health nurses were looking for during their home visits and expressed an uneasiness about feeling judged in their homes. As well, others felt that a lack of knowledge around the purpose and procedure for different trial activities, including bloodwork, may deter participation.

Environmental context and resources

Due to the various and diverse schedules of families, panellists felt that scheduling and time constraints may impact the ability of participants to join in group sessions and home visits.

Provide support to engage in the PARENT trial (TDF domain: “environment context and resources”)

To mitigate scheduling constraints for the group sessions, parents suggested offering part of the sessions through an online platform that could be accessed at their convenience, as mentioned by one parent: “maybe to have something recorded and the person can watch whenever time is convenient but it needs an opportunity to ask questions at some point (right?) – like an online course so the person can adjust, like Netflix, can see whenever you have a set time –...”. Parent participants also highlighted that availability of childcare would be very helpful at the sessions.

Application of findings

Many changes were made to the PARENT trial protocol based on the feedback of parents prior to the launch of the trial and as a way of circumventing identified barriers. Specific modifications—organized by recruitment, engagement, and intervention—are presented in Table 4. Overall, from a recruitment perspective, it was expressed that developing meaningful and authentic connections between parent participants and researchers was key to establishing a solid rapport. Providing support for parents—whether in the form of embracing various methods of involvement (virtual or in-person), reducing the number of sessions, and ensuring all forms of communications were lay friendly—were highlighted as important points for consideration moving forward. In terms of engagement, ensuring the objectives of the study were clear and that the findings were accessible and widely distributed were highlighted as important contributors to a successful trial from a participant engagement perspective. For the intervention component, ensuring that participating parents felt their involvement and contributions was being taken seriously and integrated where possible was highlighted during panel discussions. Examples include modifying research approaches (where possible/appropriate) and touching base with participating families throughout the trial.

Table 4 Resulting modifications to trial and application of findings
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Discussion and future directions

The present study provides an example of the methodological approach and preliminary engagement of parent participants as co-designers in cohort embedded RCTs in young children. Results of this study highlight the importance of gaining of parents’ feedback when designing a clinical trial in early childhood. As parents contribute valuable time to participate in the TARGet Kids! cohort study, ensuring their ideas, concerns, and priorities are included in planning and implementation is important to conducting clinical research which is most relevant for children and families. The familiarity of parents with the processes, recruitment and communication techniques of TARGet Kids!, as well as possessing good rapport with the TARGet Kids! research staff, enabled the successful involvement of parents for this study.

Developing stronger patient involvement in the organization and delivery of clinical trials is central to health service and research reform [38]; panellists in this study highlighted the importance of parents serving as co-builders in the trial and to provide suggestion on the role and expertise of individuals involved in leading and/or delivering intervention components (particularly, the home visits). This recognition reflects evidence that parents of young patients can be involved and make a difference at multiple stages of research planning and delivery [39]. In accordance with Ocloo and Matthews’ narrative review on patient and public involvement in healthcare [12], the barriers elicited from the PARENT panel fall primarily under “communication issues” (i.e. access to and understanding of information and understanding of the study), “tokenism” (i.e. symbolic effort to be inclusive to members of a particular group in order to give the appearance of equality), “poor health literacy” (i.e. difficulty understanding the trial and information required to participate), and “inadequate information about involvement” (i.e. unsure about how to contribute and whether contributions will be used/applied).

Our findings highlight the contribution of patient-oriented research from the initial stages of designing and implementing a clinical trial. In line with published work by INVOLVE (UK) [7], parent input can assist researchers with constructing and conducting trials from a logistics and process perspective (e.g. enhanced recruitment and participant buy-in, improved compliancy and study connection, and stronger retention) to ensure relevant research questions are asked. Patient engagement in studies conducted among other groups has resulted in higher recruitment and retention rates [40], better acceptability of treatment options [12], relevance of research findings [2], and improved translation of research findings into clinical practice [41]. However, long-term evaluation of patient engagement in research is not yet available, and limitations may exist such as lack of available resources to meaningfully engage patients in all aspects of research, including training, compensation, and time [12]. Patients taking part in research have highlighted the need to clearly define roles based on unique skills or expertise each patient brings to the team, and provide feedback and updates about their impact on research conduct and outcomes [42]. This has been suggested to improve the perceived value of participation to patient partners, which is key to sustained, trusting relationships between patients and researchers [42]. While this study focused on consulting parent partners for input during the planning phase of the PARENT trial, it is important to consider the spectrum of patient engagement (inform, consult, involve, collaborate and empower) [10] and continue to extend opportunities for parent participation throughout the study where possible.

Learning from this experience, we have established a Parent and Clinician Team (PACT) within TARGet Kids! to involve parents in all stages of co-design for all cohort embedded clinical trials. Both structured and unstructured discussion during semi-annual meetings allows for collaborative relationships to be built and provides space for parents to bring concerns, questions and ideas from their communities to the TARGet Kids! team. Between meetings, parents have the option to volunteer as partners for specific studies, which includes reviewing and providing feedback on grant proposals, study protocols, and procedures. On an ongoing basis, parents make recommendations for knowledge translation and dissemination strategies that are accessible and impactful to their families and communities, such as print materials, social media and email newsletters. Based on the findings from the focus group, it is believed that this approach will continue to offer support (peer or expert) and create a sense mutual respect and reciprocity between clinicians, researchers, and parent partners.

Limitations of this study include the small sample composed entirely of mothers and primarily of high socioeconomic status and European descent, thus constraining the generalizability of these findings. Additional focus group discussions may have resulted in more fruitful discussions and a wider breadth of responses. However, given that the goal of these discussions with parents was not to reach saturation, but to gain a preliminary view of the acceptability and relevance of the PARENT trial and potential barriers and facilitators to participation, the voices and opinions of seven participants were deemed sufficient to achieve the study’s goals. Because of the tight timeframe between collecting and applying participant suggestions and deploying the trial, recruiting parents and coordinating a meeting time that worked for all invitees proved challenging. Consequently, it was important for the research team to remain flexible and discuss novel ways to bring the participants together, hence the use of a virtual interviewing platform during the evening.

With the PARENT trial now underway, evaluations are being recorded to ascertain whether the suggested changes have resulted in better study logistics and improved outcomes compared to previous recruitment efforts undertaken by the TARGet Kids! research team. A second round of focus groups will take place with parent participants in the PARENT trial to elicit their feedback on their overall perspectives on the delivered protocol and recruitment methods, to gain perspectives on actual barriers/facilitators to parents’ participation, and to identify suggestions for improving parent engagement. The findings from this work will be used to further refine, adjust, and modify the next iteration of the PARENT trial, which will also serve as an opportunity to discuss the efforts made by researchers to incorporate parent suggestions and what additional steps are required for improved patient engagement.

Conclusions

The PARENT trial provided an opportunity to engage parents around an obesity prevention clinical trial embedded in a cohort study. A focus group with parents prior to the trial launch provided important insights which enhanced the study design. We hypothesize that parent engagement in the design of the PARENT trial will improve the relevance, feasibility and impact of the trial results.