Co-developing a complex transfer intervention with parents: a case study of involvement and parental experience

Abstract

Background

Research into patient and public involvement (PPI) in healthcare has gained increasing recognition over recent decades. The insights and experiences of parents of adolescents with chronic illness are valuable and can inform adolescent healthcare services, including adolescents’ transfer from pediatric to adult care. However, few studies have documented both the process and outcomes of involving parents in adolescent healthcare research as research partners. We aimed to report and evaluate the involvement of such parents in the co-development of a complex transfer intervention and to assess their experience of involvement.

Methods

In this case study, we used the ParTNerSTEPs program as an instrumental case of PPI, where participatory design was the overarching PPI methodology. To report and evaluate parental involvement, we applied the Guidance for Reporting Involvement of Patients and Public (GRIPP2) and the Patient Engagement in Research Scale (PEIRS-22). Eleven parents of adolescents with a chronic illness were involved in co-developing the transfer intervention, ParTNerSTEPs; five also participated in a collaboration group. Parents were involved through individual interviews, a World Café workshop, an online brainstorm, individual Teams meetings, and group emails. Three parents from the collaboration group participated as research partners in this case study. They contributed to the analysis of the findings and, together with the research team, formulated recommendations for future PPI.

Results

Parents preferred a combination of in-person and online methods to involve them, but emphasized that getting to know the primary researcher was essential before transitioning to online formats. Challenges were identified, particularly in preparing parents for their role in the study. Many were unfamiliar with research terminology and academic English, which at times limited their perceived ability to contribute meaningfully to the intervention’s development and act as co-authors. Nonetheless, the PEIRS-22 total score, completed by parents from the collaboration group, (mean [SD]) was 90.3 (10.8), indicating a very meaningful level of involvement.

Conclusions

A combination of in-person and online methods was associated with a very meaningful level of involvement. Tailoring involvement strategies to parents’ preferences and capacities supports meaningful and equitable participation.

Trial registration

The Regional Committees on Health Research Ethics for the Capital Region of Denmark (no. FSP 20031083).

Supplementary Information

The online version contains supplementary material available at 10.1186/s40900-026-00840-5.

Background

Patient and public involvement (PPI) in healthcare research has gained increasing recognition over recent decades and is required by some funding agencies and policymakers [1–3]. This trend is also reflected in the latest Medical Research Council (MRC) framework, which emphasizes the importance of engaging relevant stakeholders in the development, testing, evaluation, and implementation of complex interventions [4].

The rationale for incorporating PPI in research and intervention development is multifaceted and can be categorized into three motives [5, 6]. The first is a pragmatic motive: designing an intervention or product that aligns with patients’ and their families´ needs; real-world and lived experiences can enhance the research’s acceptability, value, and impact [7]. The second motive is ethical or political, asserting that patients and their families have a right to participate and contribute to research concerning their lives and circumstances. Finally, the third motive is theoretical or philosophical – patient and family involvement is essential to understanding their challenges and developing effective solutions [5, 6].

In line with the growing emphasis on PPI, new terms such as co-creation, co-development, co-design and co-production have emerged in the research field [8]. These terms, which reflect different levels of PPI, have prompted the development of various frameworks to classify the degree of engagement and the role and influence of patients in the research process [1, 9–11]. For instance, the International Association for Public Participation (IAP2) has defined and categorized PPI along a participation spectrum ranging from minimal to full involvement [9].

Traditionally, PPI in healthcare research primarily included patients [1, 12, 13], and often overlooked the needs of their families [14]. Involving the family is particularly crucial when the patient is a child or adolescent, as parents play a key role in their child’s life and treatment [15] – a role that continues into adolescence [16]. Nevertheless, two scoping reviews have highlighted a lack of research examining the level of engagement of families and strategies to foster meaningful and sustainable partnerships with key stakeholders [17, 18]. In particular, there is limited evidence on how to effectively engage parents in the research process [17, 18]. Furthermore, PPI has been criticized as a buzzword used and as a form of tokenism, often without evaluating whether, e.g., patients and parents feel genuinely involved [10, 19].

Despite the increasing use of PPI in healthcare research, there remains a lack of studies documenting both the process and the influence of involving parents. Thus, this case study covers two objectives: 1) to report and evaluate the involvement of parents of adolescents with chronic illness in co-developing a complex transfer intervention, and 2) to assess their experience of involvement in the development phase.

Methods

The case study design is particularly well-suited for situations when an in-depth, multidimensional understanding of a phenomenon is needed, such as evaluating parents’ involvement in healthcare research [20]. We therefore adopted the instrumental case study approach, using the complex transfer intervention Parents in Transition – a Nurse-led Support and Transfer Educational Program (ParTNerSTEPs) as our case [21, 22]. ParTNerSTEPs was co-developed with relevant stakeholders, including parents, and aims to support and prepare parents of adolescents (16.5–18 years) with chronic illness before and during their child’s transfer from paediatric to adult care.

Overall methodology, setting, and level of involvement

We adopted participatory design (PD) [6, 23] as the PPI methodology for involving parents in the co-development of ParTNerSTEPs. The program was designed to be implemented in the pediatric and adult nephrology, neurology, hepatology, and rheumatology outpatient clinics at Copenhagen University Hospital – Rigshospitalet, Denmark. The complexity of the intervention necessitated broad stakeholder representation [4]; therefore, multiple groups were involved in its co-development, including parents, young people (aged19-23) who had transferred to adult care, and healthcare professionals (HCPs) from both pediatric and adult outpatient clinics. This paper focused only on the involvement of parents.

Our motivation for involving parents in the co-development of ParTNerSTEPs was to ensure that the intervention addressed their needs and was relevant and acceptable, by incorporating their experiences. Our motive was pragmatic but also theoretical/philosophical, as we sought to amplify parents’ voices during the transfer period where previous interventions have focused solely on the adolescents [24, 25]. Consequently, we selected the level ‘collaborate’ on the IAP2 spectrum, which highlights active partnerships with parents in all aspects of decision making (Fig. 1) [9].

Fig. 1.

The IAP2 spectrum of public participation

Participatory design

Participatory design (PD) has its roots in action research and is based on the principle that practical problems should be solved in collaboration with those directly affected (the users) [6]. Thus, the core principle of PD is collaboration, shifting users’ roles from being passive data providers to active co-researchers with a voice in the process [23]. This study focused on the first three out of the four phases of PD, as the fourth phase concerns the evaluation phase.

1. Needs assessment – Active engagement with users to identify and analyze specific needs.

2. Ideas generation – Collaborative process to consider the identified needs and then generate ideas and prototypes with users.

3. Testing and retesting – Process involving pilot studies, reflections, and further development of prototypes before implementation.

Study population

In total, eleven parents of adolescents with chronic illness were recruited from four pediatric outpatient clinics to participate in the intervention co-development. We applied a purposive sampling strategy to achieve variation in parent sex and the adolescent’s sex, age, and transfer status. Eligible participants were parents or guardians of adolescents with a chronic illness for at least 6 months and who had transferred or would be expected to transfer to adult services. Exclusion criteria: Parents of adolescents who were not mentally or cognitively able to take on treatment responsibility, and parents who did not speak Danish.

Some parents contributed only in the first PD phase, whereas others were engaged across all three PD phases. See Fig. 2 for an overview of the parents’ involvement.

Fig. 2.

Overview of parents’ involvement in ParTNerSTEPs

Parents from the needs assessment phase were invited to participate as co-researchers by joining a collaboration group; five parents accepted (three mothers and two fathers). The group actively contributed during the co-development of the intervention to support the sustainability of the intervention in clinical practice, align it with parental needs, and mitigate potential mismatches between user perspectives and the research agenda [26]. The roles, tasks, and recognition of the collaboration group followed the European Alliance of Associations for Rheumatology (EULAR) recommendations for involving patient representatives in scientific research [26]. The parents did not receive any payment (e.g., money, gift cards, or gifts) in return for their participation.

Methods used to involve the parents in the co-development of ParTNerSTEPs

Several methods were used to involve parents in the three PD phases, see Fig. 3. Initially, we prioritized in-person meetings, but due to restrictions related to the COVID-19 pandemic (2020), we had to change to online solutions. A dotted line in the figure indicates the shift to online methods.

Fig. 3.

Methods of parental involvement and the outcomes they influenced

Individual interviews

Initially, ELT conducted individual needs assessment interviews with 11 parents of whom 73% were mothers of adolescents (aged 16–19) with chronic illness such as juvenile arthritis, autoimmune hepatitis, kidney failure and epilepsy [16]. To ensure their comfort, parents could choose whether the interview was conducted face-to-face or by telephone. The interviews were structured in three parts. Firstly, parents were asked to share their personal stories, needs, and perspectives regarding their child’s transfer to adult care. Secondly, they were asked: “If everything were possible (in terms of money and resources), what initiatives would you suggest, to support and prepare parents during their child’s transfer?” Thirdly, and last, to avoid influencing the parents’ input, initiatives identified in previous studies were presented, and parents rated their relevance. The most commonly preferred initiatives formed the basis of the intervention [16]. Parents also advised on when, during the transition period, the intervention should be offered. Finally, all parents were invited to participate in further development through a workshop; six out of 11 agreed.

World Café workshop

We conducted a World Café workshop, guided by Clemensen et al.’s four participatory design principles: 1) give everyone a voice by creating room for visions, 2) understand and participate in the clinical practice, 3) create a learning environment for everyone, and 4) find a mutual language [6]. The World Café format created a relaxed and creative environment that allowed for diverse expression (verbal, written, or drawn) [27, 28]. The workshop was conducted using three tables, each dedicated to one of the three selected initiatives: website, educational events, and transfer consultations. Six parents, two young people (aged 19–23), and six HCPs participated. They were divided into three groups and rotated between tables every 20 minutes, ensuring that all participants discussed all three intervention initiatives.

Before the table discussions, ELT presented findings from the individual interviews to validate the parents’ experiences and needs. The participants were asked to dream big and be creative but were informed that not everything could be accomplished due to resource limitations and regulations. Each table was facilitated and structured around reflection, content, and design questions. The reflection questions (e.g., “What makes a website good?”) helped establish a shared language and reduce professional hierarchies. To address potential power imbalances, parents and young people were not grouped with HCPs whom they knew, and participants were reassured that their contributions would not affect their child’s or their own treatment or care. Familiarity with the researcher and early validation of parental perspectives likely supported openness and reduced the power imbalance. Full workshop details are reported elsewhere [21, 22].

Online brainstorming session

ELT conducted an online brainstorming session with three out of the five parents, during which the parents were asked to reflect on how the intervention might affect parents and their children, and to suggest potential changes it could lead to. Their suggestions were discussed and visually organized into joint virtual mind maps created in real time by ELT. The research group subsequently used the mind maps to identify and select relevant outcomes for the evaluation of ParTNerSTEPs.

Individual online microsoft teams meetings

ELT held several individual online meetings with the five parents from the collaboration group using Microsoft Teams. Each meeting had a specific task, such as providing feedback on the translation of questionnaires (face and content validation) or offering advice and ideas on how to recruit participants in the RCT. Each meeting began with a brief check-in, during which daily life issues were discussed, followed by an overview of the meeting’s purpose.

Group email discussions

ELT sent several group emails inviting the parents from the collaboration group to participate in the testing and re-testing phase. They were, e.g., asked to provide feedback on aspects such as the website’s design, navigation, and ease of locating specific information. The group email discussions also aimed to keep parents informed and update them, so they continued to feel involved. Involvement in the various tasks was voluntary, and parents were free to abstain if they lacked the time or energy to participate. Consequently, only those who responded received follow-up communication.

Data collection and materials

Regarding the first objective, we used the Guidance for Reporting Involvement of Patients and Public (GRIPP2) [29] to guide the reporting of parental involvement in the co-development of ParTNerSTEPs. GRIPP2 was developed by Staniszewska et al. to improve the quality, consistency, and transparency of reporting PPI. It includes two versions: a long form (GRIPP2-LF) and a short form (GRIPP2-SF). We used GRIPP2-SF, which is divided into five sections: 1) Aim, 2) Methods, 3) Results, 4) Discussions, and 5) Reflections, and include both positive and negative aspects of PPI. Data were written descriptions and extracts from logbooks from the development phase and previous publications [16, 21, 22].

To meet the second objective, we used the questionnaire Patient Engagement in Research Scale (PEIRS-22) [19, 30] to evaluate the parents’ subjective experiences and quality of their involvement in the co-development of ParTNerSTEPs. PEIRS-22 was developed and validated by Hamilton et al. [19, 30] to measure the degree of meaningful involvement of patients and family caregivers in research. PEIRS-22 assessed participants’ subjective perceptions of their involvement using 22 items that measured the planned, encouraged, and valued participation of patients and caregivers. Each item is rated on a 5-point Likert scale, ranging from ‘strongly disagree’ to ‘strongly agree’, with higher scores indicating a higher level of meaningful engagement [19, 30]. We used the Danish version, validated by Christiansen et al. in 2023 [31]. Only parents in the collaboration group (n = 5) were invited to complete the questionnaire, via the online platform REDCap, in September-October 2024. All responses were collected anonymously to ensure confidentiality.

All data were stored in accordance with the Danish Data Protection Agency (no. p-2020–550).

Data analysis

Written descriptions of the five GRIPP2 sections were analyzed in a workshop in July 2025. The five parents from the collaboration group were invited to be co-authors of this paper and participate in the workshop alongside the primary investigator (ELT) and KB, who both were involved in the co-development of the intervention. Three parents, all mothers (STC, HGS, NO) accepted the invitation and were subsequently referred to as parent research partners (PRPs). The two fathers never replied. Before the workshop, descriptions of the various methods used to involve the parents in the co-development of the intervention was translated into Danish and sent to the PRPs, so they could prepare. During the workshop, ELT presented the methods and facilitated an open discussion, in Danish, on the positive and negative aspects of PPI in the co-development of ParTNerSTEPs (see photos). The PRPs’ sticky notes and oral feedback were digitally recorded, transcribed, and subsequently analyzed deductively by ELT and reported under each of the five GRIPP2-SF sections, with approval from the entire author group, including the PRPs. A detailed report of the PPI process during the workshop and co-author of this paper is provided in Additional file 2.

Photo 1 and 2.

Written consent to use the images has been obtained from all participants in the workshop

Responses from the PEIRS-22 were scored using a 5-point Likert scale, with each item assigned a numerical value ranging from 0 (strongly disagree) to 4 (strongly agree). The total PEIRS-22 score was calculated by summing all item scores, dividing by 88 (22 items × 4), and multiplying by 100 to obtain a standardized percentage score. Based on the thresholds defined by Hamilton et al. [30, 32], a total mean score of 92 or higher reflects extremely high meaningful involvement, whereas a score of 70.1 or below indicates a low to moderate level of meaningful involvement [30, 32]. PEIRS-22 scores were also presented and discussed by the PRPs during the workshop.

Results

Evaluation of the involvement of parents during the co-development of ParTNerSTEPs

GRIPP2-SF provides an overview of how parents were involved throughout the co-development of ParTNerSTEPs (Table 1). A detailed presentation of positive and negative outcome of involving parents and the various methods can be found below.

Table 1.

Involvement of parents in the co-development of ParTNerSTEPs using GRIPP2-SF

Section and topics	Item
1: Aim Report the aim of PPI the study	To involve parents in the co-development of a complex intervention targeting parents of adolescents with chronic illness, to ensure that the intervention addressed their needs and was relevant and acceptable.
2: Methods Provide a clear description of the methods used for PPI in the study	The overall method used for involving the parents was participatory design (PD). However, several methods (interviews, workshop, online brainstorm/mind maps, online Teams meetings and group emails) were used to involve the parents in the different PD phases. An overview of the methods is provided in Sect. “Methods used to involve the parents in the co-development of ParTNerSTEPs” and Fig. 3.
3: Results Outcome – Report the results of PPI in the study, including positive and negative outcomes	Parents influenced several outcomes in the co-development of ParTNerSTEPs, including: - The selection of intervention initiatives (Website, educational events, and transfer consultations) was chosen by the parents. - The decision to offer the ParTNerSTEPs program to parents ½-1 year before their child’s transfer at age 18, rather than earlier, as initially planned. For recruitment reasons we offered ParTNerSTEPs to parents of adolescents aged 16.5–17.5. - The content of the three initiatives, e.g., that the website and educational events should also focus on the transfer within the municipal system and provide educational support. Additionally, the transfer consultations should include a farewell consultation in pediatrics. - Parents’ input on possible outcomes determined the choice of questionnaires and focus areas in the evaluation phase. - The graphic design of the website was decided exclusively by the parents (http://www.partner-steps.dk). - The parents reviewed the wording of the questionnaires and recruitment materials to ensure that the language was understandable and relevant to other parents. - Co-authorship of the protocol paper was challenging for the parents due to unfamiliarity with academic English, resulting in less contribution to the formulation and review of the manuscript.
4: Discussion Outcome – Comment on the extent to which PPI influenced the study overall. Describe positive and negative effects	The parents had a significant influence on the co-development of ParTNerSTEPs, where, throughout the entire process, their voice carried the most weight in decisions that did not concern resources or the General Data Protection Regulation (GDPR). The Parent Research Partners (PRPs), therefore, assessed that the degree of involvement was somewhere between ‘involved’ and ‘collaborative’ on IAP2‘s involvement spectrum, depending on the method used:
		Positive aspects	Aspects to improve
	Interviews	Building trust and a safe setting for sharing sensitive experiences	Prompts to help parents generate ideas can be helpful.
	Workshop	Open and honest discussions with mutual respect. A free discussion, then parents and staff did not know each other.	Unclear instructions and dominant participants could prevent some from contributing, which is why a strong facilitator is needed.
	Online brainstorming	Shared mind map supported reflection and mutual understanding.	Preparatory material could be sent in advance. An in-person format could be beneficial for abstract tasks.
	Online Teams meetings	Flexible scheduling and no travel time. Time-efficient, then focused meetings on specific tasks.	Limited group cohesion and sense of community.
	Group emails	Gave a clear overview of the process and upcoming tasks. Strengthened the sense of being in a group and allowed time for reflection before responding.	Lack of follow-up with non-responders led to exclusion and uncertainty about continued involvement.
5: Reflections Critical perspective -Comment critically on the study, reflecting on the things that went well and those that did not, so others can learn from this experience	This study demonstrated that it is possible to involve parents in the development of a complex intervention by combining in-person and online methods. The flexible format was well-received, though participants stated that a trusting relationship with the primary researcher was essential before transitioning to virtual meetings. While the online format allowed parents greater autonomy in scheduling, it also reduced their sense of belonging to a group. However, they did not express a need to be part of a larger research team, as they felt their perspectives were represented and carried forward by the primary researcher, who acted as a bridge between the parent group and the research team. The study also revealed challenges in adequately preparing parents for their roles. Many lacked familiarities with research concepts, academic English, and the expectations associated with participation. This often led to confusion, especially at the beginning of a new task. PRPs expressed a need for clear, accessible introductions and preparatory materials—preferably in their native language—to enable more meaningful engagement. These reflections underscore the importance of tailoring involvement to the needs, preferences, and capacities of parent participants to foster authentic involvement.

PPI = Patient and Public Involvement, PRPs = Parent Research Partners

Results influenced by the parents

The parents had a significant influence over the selection of initiatives that the intervention should include, with the majority preferring an informative website, educational events, and transfer consultations. The parents also had a significant impact on shaping the content and format of the intervention. The insights gathered from the workshop were instrumental in designing and refining the three core components, based on the parents’ suggestions. However, not all suggestions could be accommodated. For instance, parents proposed creating an interactive forum within the website to facilitate peer support and sharing of experiences, but this was not feasible due to General Data Protection Regulation (GDPR) constraints. Additionally, while many parents valued the opportunity for in-person educational events, COVID-19 restrictions (2020) made this impossible during the development phase, necessitating a shift to an online format.

The parents’ feedback also guided the timing of the intervention. Initially, the research team had planned to offer the ParTNerSTEPs intervention to parents of adolescents aged 15. However, parents strongly recommended that we adjusted the timing to 6–12 months before the adolescent’s transfer to adult care at age 18. They felt that offering the intervention earlier would be premature, as parents were not yet emotionally prepared to focus on transfer-related issues at that stage. Based on this feedback, and considering practical recruitment needs, we decided to target parents of adolescents aged 16.5–17.5 years.

Two parents agreed to co-author the intervention protocol paper. Despite their commitment, they described difficulties engaging with the academic English of the manuscript, which limited their feeling of being able to contribute. Several noted that a Danish translation of the text and an in-person meeting to review and discuss the content would have facilitated deeper involvement and more meaningful input.

Advantages and disadvantages of the used methods

Interviews: The individual interviews gave ELT insight into the complex and varied roles, perspectives, and needs of parents. PRPs appreciated being heard and taken seriously and valued the chance to influence the intervention. They welcomed the choice of interview format and described the setting as safe for sharing personal and sensitive experiences. Several highlighted that the interviews helped build trust and a working relationship with ELT, which supported their involvement later in the process. Some parents from the interview found it challenging to suggest new intervention ideas but found it easier to respond to and validate initiatives identified through the literature.

Workshop: PRPs found the workshop to be a space for open and honest discussion, where they felt, their views were listened to and taken seriously by the HCPs. They valued the shared language that developed across specialties and were inspired by hearing others’ perspectives. Being placed with staff who were not directly involved in their child’s care made it easier to speak freely. However, the beginning was somewhat confusing due to unclear instructions, and some participants spoke a lot, which made it harder for others to join the conversation without support from a strong facilitator. A few PRPs also found it emotionally difficult to discuss challenging topics, which may have deterred some from participating further.

Online brainstorming sessions: PRPs initially found the task unclear and felt that a presentation or written material sent in advance would have helped them prepare for the brainstorming session. Over time, the purpose became clearer, and they appreciated the opportunity to exchange ideas and reflect on how ParTNerSTEPs might benefit other adolescents and their parents. The shared mind map helped check mutual understanding. Some suggested that the brainstorming process might work better in an in-person setting.

Online Teams meetings: PRPs appreciated the individual online meetings, as scheduling was easier with only two parties and no travel time was required. They found the format to be time-efficient and valued the regular, focused meetings on specific tasks related to ParTNerSTEPs. While the format worked well due to their familiarity with ELT and the project, they noted a lack of group cohesion and sense of community. The research group felt well-informed through continuous updates from the individual meetings and did not see a need for larger joint sessions.

Group email discussions: PRPs found that group email discussions provided a helpful overview of the process and upcoming tasks and receiving them as a group strengthened their sense of belonging. The PRPs appreciated the emails, as they made them feel that their input remained valued for the project. Some of the PRPs felt pressure to respond quickly out of concern they might forget, while others welcomed the time to reflect before replying. However, parents who did not respond received no follow-up, leaving them uninformed and making it unclear whether they lacked time or no longer wished to be involved.

Parents’ experiences of involvement

All five parents in the collaboration group completed the PEIRS-2 questionnaire. The mean PEIRS-22 total score (SD) was 90.3 (10.8), indicating a very meaningful level of involvement. Table 2 presents the mean score for each item.

Table 2.

The parents’ mean score for each PEIRS-22 item (N = 5)

	Mean score (min-max)
Procedural Requirements - general experiences throughout the project
The research team members were properly introduced to each other	2.8 [1–4]
In general, I had sufficient opportunities to contribute to the project	3.6 [3, 4]
I was able to perform my tasks for the project	3.6 [3, 4]
I participated in making decisions about the project	2.6 [2, 3]
I received sufficient updates about the project	3.8 [3, 4]
Communication within the research team was clear throughout the project	3.4 [2–4]
The project was worth the time I spent on it	4.0 [4]
Convenience
I had the opportunity to provide input into selecting my tasks for the project	3.2 [2–4]
Throughout the project, I had sufficient time to complete my tasks for the project	3.8 [3, 4]
I had opportunities to express my views	4.0 [4]
Contributions
I contributed by providing my perspective	3.4 [3, 4]
My contributions were a good use of my time	3.8 [3, 4]
My workload in the project was manageable	3.8 [3, 4]
Team Environment and Interaction
I was an equal partner in the research project team	3.0 [2–4]
There was trust among the research project team members	3.2 [2–4]
Support
I received sufficient support to contribute to the project (e.g., orientation, readings, training workshops, webinars)	3.2 [2–4]
Any concerns I had were addressed	3.8 [3, 4]
Feel Valued
The research project team appreciated my contributions	3.8 [3, 4]
I was offered sufficient recognition for my contributions (e.g., payment, authorship, or gifts)	3.0 [2–4]
Benefits
I enjoyed being a part of the project	3.4 [2–4]
I made an impact on the decisions in the project	3.4 [2–4]
My involvement had positive impacts on my life	3.4 [2–4]

0 (strongly disagree) to 4 (strongly agree)

Eight items received either the highest or second-highest possible ratings from all parents (3.8–4.0), including statements such as “I had opportunities to express my views” and “The research project team appreciated my contributions”. The lowest-rated item, “I participated in making decisions about the project,” received a score of 2.6. The lower score was discussed by the PRPs, who noted that they did not perceive ParTNerSTEPs as their project; therefore, they felt that it was appropriate for final decisions to rest with the research team. Accordingly, they assessed their role on the IAP2 spectrum as “involve” or “collaborate” rather than “empower” (Fig. 1), where decision-making authority lies with the public.

Discussion

In this case study, parents preferred a combination of in-person and online methods when co-developing the complex transfer intervention, ParTNerSTEPs. We found that the GRIPP2-SF checklist and the PEIRS-22 scale are useful tools for reporting and evaluating parental involvement in healthcare research. Parents involved in ParTNerSTEPs reported a total PEIRS-22 score indicating very meaningful involvement, comparable to levels reported in other studies [31, 33]. The highest-rated items, such as “I had opportunities to express my views” and “The research project team appreciated my contributions”, indicate that parents felt heard and influential in shaping the intervention. When reflecting on the methods used to involve parents in ParTNerSTEPs, PRPs emphasized that both individual interviews and the workshop provided safe spaces in which they could speak freely and felt listened to, which may explain these high scores. The PRPs’ experiences contrast with findings from a systematic review by Malterud & Elvbakken [34], which highlights the risk that patients or public contributors may not have real influence on decisions in research projects. In addition, the results from GRIPP2-SF revealed specific examples of how parents influenced the content and timing of the intervention, e.g., a later start of the program than initially planned by the research team. The PEIRS-22 items receiving some of the lowest scores were “The research team members were properly introduced to each other“and “I was an equal partner in the research project team”, suggesting that PRPs did not fully experience being part of a research team. This is supported by the GRIPP2 data, which show that PRP reported a strong personal connection with the primary investigator but also noted that methods, such as individual online Teams meetings, contributed to feelings of alienation from the group. In contrast, group emails were described as fostering a sense of community. To strengthen team cohesion, group-based activities, such as group meetings or emails, should therefore be prioritized; however, this approach may affect parental participation due to increased logistical demands when multiple parties are involved.

The co-development of the ParTNerSTEPs program spanned four years (2019–2022), from the initial needs assessment interviews to the publication of the intervention protocol. The extended timeframe made it challenging to sustain consistent parental engagement, and we observed a decline in participation from some parents over time. All parents continued to receive regular emails with updates and new tasks. However, the primary researcher did not follow up to discuss the option of stepping back temporarily or adjusting level of involvement. The lack of follow-up contrasts with recent recommendations for fostering meaningful engagement, which emphasize the importance of regular check-ins and ongoing dialogue about contributors’ roles and expectations [32, 35]. One tool proposed to support such discussions is the Involvement Matrix, developed by Smits et al. [11], which presents various roles, ranging from listener to decision maker, that patients and parents can select across various project phases. While our PRPs welcomed regular informal check-ins, they found the matrix too academic and overwhelming for practical use.

Our findings also suggest that sustained involvement may be supported by transitioning to online solutions after an initial period of building a trustworthy relationship with the primary investigator. Once the relationship was established, the PRPs appreciated the flexibility and autonomy provided by the online format, which saved time and made scheduling easier. This aligns with findings by Oliver et al. [36], who identify time, cost, and travel as significant barriers to in-person involvement in co-produced research. However, a study involving adolescents and young adults found that in-person meetings were preferred [37]. This may suggest that adolescents with chronic illnesses can feel alienated from their friends and seek peer connections and a social community through involvement in projects [38, 39]. In contrast, parents often have jobs, caregiving responsibilities, other children, older parents, and limited time, making online formats more feasible for their participation. Together, these findings suggest that PPI in transition/transfer-focused healthcare research should differentiate involvement methods for adolescents and parents, as their needs and preferences are not necessarily aligned.

Our results also revealed challenges in adequately preparing and supporting parents as co-researchers. Several PRPs found it difficult to understand the various tasks at the outset and wished they had received written materials in advance, translated into their native language. The two parents who chose to be co-authors of the protocol paper also struggled with academic English and felt unable to contribute fully. A lack of scientific or perceived academic competence is a well-documented barrier to meaningful involvement [18, 34, 35]. Based on these challenges, the PRPs suggested that more abstract and academic tasks – such as outcome selections and co-authoring – should be facilitated through in-person meetings, with translated materials and ongoing support to help them carry out their role. This aligns with recent recommendations stating the importance of supporting patient and public contributors in developing their skills. Wang et al. [32] recommend that researchers create opportunities to build relevant competencies in patient partners. Baba et al. [35] identify six key enablers of meaningful engagement, including tailored training for specific project activities and easy access to core materials, such as glossaries and slide decks. The applicability of the International Committee of Medical Journal Editors authorship criteria (the Vancouver guideline) in the context of PPI can be debated. The guideline states that co-authors must be accountable for all aspects of the work [40], which may be unrealistic for patient and public contributors who often lack formal research training. We propose that PPI contributors can be legitimately recognized as co-authors if they have been involved throughout the research process, have accepted the article’s main findings, have been taken through the abstract in layman’s language, and have contributed to the discussion section at a brainstorming level. Ultimately, it is also worth considering whether PPI representatives could assume a more prominent role in the national dissemination of research findings and advocation for key changes.

This study also had limitations, as only mothers participated as co-authors of this paper. We observed that the two fathers became less involved throughout the ParTNerSTEPs development, suggesting that our results may be shaped primarily by maternal perspectives. However, both fathers did complete the PEIRS-22 questionnaire. The overrepresentation of women in PPI is a well-documented limitation in research [36, 41]. One explanation could be that women often lead PPI work; and the approaches used may therefore be more appealing or accessible to them [36]. Another explanation is that mothers are often the primary caregivers in families of children with chronic conditions [42], which may make them more inclined to participate in research related to their child. Future research should therefore explore how male participants prefer to be involved in research projects.

Recommendations for further research

Based on the results of this case study, we recommend that future research projects involving parents actively engage them, as they offer valuable insights and guidance for developing interventions tailored to their specific needs.

Our findings suggest that individual interviews, workshops, and online methods, such as virtual mind maps, online meetings, and group email discussions are suitable and feasible formats for promoting parental involvement. To support meaningful and sustained engagement, we suggest that future research consider the following:

• Offer a flexible format that combines in-person and online methods, introduced after a trusting relationship has been established between the researcher and the parent group.

• Maintain an ongoing dialogue with parents about their preferred level of involvement throughout the research process; they do not necessarily have to be involved in all phases.

• Translate all research materials into the parents’ native language to enhance accessibility and comprehension.

• When co-authoring papers, facilitate in-person workgroups where parents can provide oral input instead of written feedback.

• Prepare parents adequately by sending written materials (including specific questions or tasks) in advance, in their native language.

• Consider developing alternative methods to support the involvement of fathers, who may prefer different formats or modes of engagement.

Conclusion

A combination of in-person and online methods was associated with a very meaningful level of involvement. Our findings underscore the importance of preparing parents for their role in research projects and tailoring methods to their preferences and capacities, thereby supporting meaningful and equitable participation.

Electronic supplementary material

Below is the link to the electronic supplementary material.

Abbreviations

BAE

Bente Appel Esbensen

ELT

Ena Lindhart Thomsen - author

EULAR

European Alliance of Associations for Rheumatology

GDPR

General Data Protection Regulation

GRIPP2

Guidance for Reporting Involvement of Patients and Public

GRIPP2-LF

Guidance for Reporting Involvement of Patients and Public – Long Form

GRIPP2-SF

Guidance for Reporting Involvement of Patients and Public – Short Form

HCPs

Healthcare Professionals

HH

Helena Hansson

HGS

Heidi-Christina V Grabow Scheelhardt – parent research partner

IAP2

The International Association for Public Participation

KB

Kirsten Boisen - author

MRC

Medical Research Council

NO

Nina Olsen – parent research partner

ParTNerSTEPs

Parents in Transition – a Nurse-led Support and Transfer Educational Program

PD

Participatory Design

PEIRS-22

Patient Engagement in Research Scale

PPI

Patient and Public Involvement

PRPs

Parent Research Partners

RCT

Randomized Controlled Trial

STC

Susanne Thing Christensen – parent research partner

Author contributions

All authors contributed to the design and delivery of the study. ELT, KB, BAE, and HH planned and prepared the study. ELT, KB, HGS, NO, and ST participated in the workshop, evaluating and analyzing the data. All authors contributed to the follow-up discussion of the results and recommendations. ELT, KB, BAE, and HH drafted the initial manuscript. HGS, NO, and ST significantly supported the drafting of the manuscript. All authors have contributed to writing the manuscript and have read and approved the final version.

Funding

Open access funding provided by Copenhagen University. This project is funded by Danish Regions and the Danish Health Confederation Development and Research Fund (project 2724). The foundation did not have any role in the design or conduct the study.

Data availability

The datasets generated during and/or analyzed during the current study (transcription of the workshop and PEIRS-22 data) are not publicly available but can be made available upon reasonable request to the first author (ELT).

Declarations

Ethics approval and consent to participate

The study was approved by the Danish Data Protection Agency (no. p-2020–550) and the Regional Committees on Health Research Ethics for the Capital Region of Denmark (no. FSP 20,031,083). Data were anonymized and managed confidentially. Members of the collaboration group received information about their role as co-researchers and signed a confidentiality agreement (Additional file 1).

Consent for publication

Not applicable.

Competing interests

The authors declare no competing interests.