1 INTRODUCTION

Young people with a chronic condition (YPCC) generally play a passive role in research. They are invited to participate in questionnaires and interviews. There is, however, growing consensus that YPCC should be actively involved in research that concerns them. This is also termed ‘Patient and Public Involvement’, or PPI. Hart defined PPI of young people as ‘the process of sharing decisions which affect one's life and the life of the community in which one lives’.1 INVOLVE—the former PPI advisory group of the British National Institute for Health Research, now replaced by the Centre for Engagement and Dissemination—devised a general and more practical definition: PPI is about ‘research being carried out “with” or “by” members of the public, rather than “to”, “about” or “for” them’.2

Many researchers and YPCC are currently struggling with how to do PPI in research.3 PPI can take many forms, with YPCC being involved in various ways and in several stages of various types of research.4, 5 The literature on PPI of YPCC provides a plethora of examples, from intensive partnerships with a few YPCC6-8 to consultations with advisory panels.9, 10 The flexibility of PPI makes it possible to adapt it to the research and the people involved.11 At the same time, it complicates PPI practice, since there is no ‘blueprint’ for doing it right.5, 12 PPI with YPCC is further complicated by the tendency to view YPCC as vulnerable and inexperienced and to underestimate their competence as decision-makers.1, 3, 6, 12 This exacerbates the general issue of power imbalances in PPI.3

In recent years, there has been a rising demand for demonstrating the impact of PPI. The literature on PPI of YPCC shows that impact can take many forms.4, 5, 11, 13 It is suggested that PPI can increase the relevance and quality of research. YPCC are able to provide new insights from their lived experience, which can improve aspects such as research design,14, 15 data collection6, 14 and dissemination of results.14, 16 PPI is also thought to have a positive impact on the personal development of the YPCC involved, since they learn new skills and gain more self-confidence.14, 17 Among researchers, PPI can increase their commitment to research16 and evoke feelings of inspiration and pride.14 Finally, some research suggests that PPI can enhance the position of all YPCC in society by promoting their right to be heard and supporting inclusivity.14, 18

Demonstrating the impact of PPI of YPCC can help resolve some of the complexities of PPI.5 It provides insight into the achievements of PPI. In addition, demonstrating impact in relation to specific PPI approaches can help identify the PPI approaches that are most likely to realize these achievements. In the words of Staniszewska and colleagues: ‘it requires evidence to inform best practice’.19 However, best practice can solely be informed by evidence that is robust and of high quality. Researchers in the field of PPI are currently struggling to find ways to conduct robust evaluations of PPI of YPCC. Several literature reviews—published during a time span of 16 years—have shown that the current evidence base on PPI of YPCC and its impact is weak.4, 11, 20 In recent decades, limited progress has been made to change this.4

This limited progress is partly the result of poor understanding of what meaningful impact exactly entails.21 A previous study reported that researchers and YPCC find it difficult to specify the impact they achieved beyond general descriptions.22 A more detailed understanding of impact can extend our knowledge about the difference that PPI can make and which approaches are effective. We therefore conducted a systematic and extensive study of the perspectives of researchers and YPCC on meaningful impact. We addressed the following research question: From the perspective of researchers and YPCC, what is considered meaningful impact of PPI in research?

2 MATERIALS AND METHODS 2.1 Design

Q methodology combines qualitative and quantitative techniques to systematically study how people think about a certain topic.23-25 In Q methodology, between 40 and 60 participants are presented with a sample of 20–100 statements, which they rank order onto a grid. After this, they are asked to reflect on the choices they made. Factor analysis is then conducted to reveal patterns of similarity in how statements were sorted by respondents. Unlike conventional factor analysis, in Q methodology, the individual rankings—not the different statements—are taken as variables. The resulting factors represent groups of individuals with similar perspectives. Participants’ reflections on their rankings are used to interpret and describe the factors.

We chose Q methodology because it was very suitable to answer our research question for several reasons. First, it can be used to systematically study perspectives and compare their similarities and differences.24, 25 Second, it also prompts participants to carefully weigh the importance of various statements, as the method compels them to make a choice.24, 26 Third, it combines the strengths of quantitative and qualitative research. The factors retrieved in quantitative analyses are given meaning by using participants’ reflections on their sorting of statements.24, 25

2.2 Participants

The participants in the study were researchers and YPCC between 15 and 30 years old. Participants were required to have at least some experience with PPI of YPCC. Researchers were recruited through the authors’ networks in the academic field. YPCC were recruited through the network of an adolescent with a chronic condition who was involved in the preparation of this study and an announcement placed on the website of JongPIT, a Dutch foundation for and led by YPCC. Snowballing was used to contact additional researchers and YPCC.

After recruitment, 50 participants sorted statements and were interviewed. However, four participants were excluded from the analysis. During data collection, it became clear that one researcher and one YPCC had not fully understood the sorting task. Two YPCC were not familiar with PPI. Consequently, the final sample consisted of 46 participants: 20 YPCC and 26 researchers. All researchers and YPCC were Dutch and therefore mostly had experience with PPI in the Netherlands. There were two exceptions: one Dutch researcher conducted research in Denmark and another in Canada. Researchers were not reimbursed for their participation in the study; YPCC received a gift certificate.

2.3 Data collection

Statements were collected about various types of impact, such us ‘Young people acquire new knowledge and skills’ and ‘PPI contributes to a society in which everyone can participate’. A recently conducted literature review was used as the primary input to formulate statements4 by scanning the data on reported motivations and benefits of PPI. This was performed by the first author. During this process, she merged some statements that were very similar. This resulted in a list of 39 statements, which was discussed by all authors and the adolescent with a chronic condition who was involved in the preparation of this study. In an iterative process, it was decided to remove some additional statements to eliminate repetition. In addition, statements were clarified and shortened to improve comprehensibility. The final selection (also called the Q sample) consisted of 33 statements (Table 2).

Participants were asked to rank order the Q sample using a Q sort table (Figure 1). Due to the COVID-19 circumstances, they were invited to do this digitally. Participants were sent a link to the ranking exercise, which was programmed using the VQMethod.27 The ranking exercise started with an overview of all 33 statements. First, they were asked to sort the statements (options: agree, neutral, disagree) guided by the question ‘What are your motivations for doing PPI?’ The second step was to rank all statements in the Q sort table. Based on the first sorting exercise, participants were instructed to rank the statements based on how much they agreed with them. The more they agreed with a statement, the more they placed it to the right of the Q sort table. The more they disagreed with a statement, the more they placed it to the left. The statements they agreed or disagreed with most were placed at the extreme right (+4) and left (−4). After finishing the Q sort table, participants were asked to answer a few short questions about their background and previous experience with PPI of YPCC (‘little’, ‘some’ or ‘much’).

Q sort table

After the ranking exercise, telephone interviews or video calls were conducted with participants to reflect on their individual ranking of statements. Participants answered questions about the two or three statements that they placed at the extreme right (+3 and +4) and left (−3 and −4) sides. Some additional questions about successful PPI and impact were included to ensure that all relevant topics regarding meaningful impact were addressed (Box ). All interviews lasted a maximum of 45 min. They were audio-taped and transcribed.

2.4 Analysis and interpretation

The individual rankings of statements—also termed Q sorts—were analysed using principal component analysis, followed by Varimax rotation. Analyses were conducted in Stata 15.0 using qfactor.28 The selection of a factor solution was based on both quantitative and qualitative criteria.23, 29 Some statistical features were examined. For example, only factors with an eigenvalue in excess of 1.00 were selected. Another requirement was that at least two Q sorts must load onto each factor. Ideally, the percentage of explained variance in the chosen factor solution is 35%–40% or higher.29 In our analyses, factor solutions with 2–7 factors fulfilled these requirements.

Since factors should be interpretable and represent coherent and comprehensible narratives, the final factor solution was based on the qualitative interviews.29 As a part of Q methodology, the views expressed during the interviews were compared with the idealized Q sort of each factor, which are called the composite sorts. In a composite sort, ‘the Q sorts of all participants who define a given factor are merged together to yield a single (factor exemplifying) Q sort’.23 The choice for the final factor solution was based on the extent to which composite sorts were consistent with the qualitative reflections of the participants whose Q sorts defined each factor.

Interviews were analysed in MaxQDA. The statements of the ranking exercise were translated into codes. All fragments reflecting on specific statements were coded within the corresponding code. This resulted in an overview of how participants interpreted different statements and how they reflected on them. The principal author compared the interpretations and reflections of participants and assessed the extent to which they represented similar perspectives on impact. Based on this, a preliminary factor solution was chosen, which was discussed with the other authors. The discussion focused on whether the factor solution covered all perspectives on impact and how they related to each other. Ultimately, consensus was reached on the definitive factor solution.

Interpretation of the factor solution was based on the expressed views and elaborations in the interviews and the relative placement of all statements within each composite sort. Particular attention was paid to characterizing and distinguishing statements. Characterizing statements are statements on or near the extremities of the Q sort table (+4, +3, −3 and −4). Distinguishing statements are statements that are placed on significantly different positions in the Q sort tables of different composite sorts.

2.5 Validity

All participants provided informed consent before they started the ranking exercise. The study was conducted in accordance with the General Data Protection Regulation. Formal ethical approval of this study was not required under the prevailing Dutch legislation.

3 RESULTS 3.1 Description of participants

Table 1 presents a summary of the characteristics of the study participants. On average, YPCC were 23 years old (range 17–29 years). Their experience with PPI varied from little or some (50%) to much (50%). The researchers who participated were on average 39 years (range 22–63) of age. Their experience in doing research varied, with interns, PhD students, postdocs, senior researchers and professors taking part in the study. They had little or some (62%) or much (38%) experience with PPI of YPCC.

Table 1. Study participants Young people with a chronic condition Researchers N % or M (min–max) N % or M (min–max) Total 20 100 26 100 Sex Female 12 60 23 88 Male 8 40 3 12 Age (years) 20 23 (17–29) 26 39 (22–63) Educationa Elementary school 0 0 0 0 Secondary education 3 15 0 0 Postsecondary education 17 85 26 100 Experienced in PPI of young people with a chronic condition (%) Little or some experience 10 50 16 62 Much experience 10 50 10 38 Abbreviation: PPI, Patient and Public Involvement. 3.2 Perspectives on meaningful impact

Four factors and composite sorts were extracted from the data, that is four distinct perspectives on meaningful impact of involving YPCC in research. These factors were defined by 37 Q sorts (80%); 9 Q sorts did not load significantly onto any of the factors. Table 2 shows the position of each statement in the composite sorts.

Table 2. Statements (Q sample) and their position in the four composite sorts Factor A Factor B Factor C Factor D 1 Research involving young people receives more media attention −3a −3a −2a −4a 2 Young people help researchers to better understand research outcomes 1 4a −1 0 3 Young people describe research outcome in a language young people understand 1 1 0a 1 4 When a peer describes research, young people can make a more informed decision about research participation 0 0 −2 −1 5 It improves the usefulness of research outcomes in practice 3 2 3 1a 6 Researchers become more creative 1 1 2 0a 7 It is useful for young people's resume −2a −2a 1 0 8 It motivates young people to participate in democracy −1a −4a −3a 2a 9 It is increasingly a requirement of funders and scientific journals that young people are involved in research −4 −1 −1 −3 10 Young people feel heard 0 3 0 3 11 It increases equality between young people and researchers 0 −2a −3a 0 12 Young people are becoming more interested in research −1 −1 −1 0 13 Young people gain new insights on themselves −1 −1 1a 0 14 Researchers obtain new insights from young people 2 3 2 1a 15 Young people get to know other young people −3 0a 0a −3 16 Young people enjoy their involvement −2a 1a 0a 3a 17 Researchers enjoy young people's involvement −1 1a −2 −1 18 It contributes to a society in which everyone can participate 0a −3a 4a 2a 19 Young people build a network −2 −2 1a −1a 20 Young people are given the opportunity to do something useful for other young people 0a 1a 2 2 21 Young people know which research methods suit the target group 1a −1 −4a −2 22 Young people come up with research questions that are important to young people 4a 2 0a 1 23 Young people put young people who participate in the study at ease 0 0 −1 −1 24 Researchers learn who exactly their research is about 2 2 2 1 25 Young people have the right to participate in research that concerns them 2a 0a 3a 4a 26 Young people and researchers build valuable relationships −1 0 0 1 27 It helps young people to think about their future −2 −2 −1a −2 28 Young people acquire new knowledge and skills 0a 2 1 2 29 Researchers look beyond just their own experiences 3a 0 1 −1a 30 Young people spread the results of research more easily among young people 1a 0 −2 −2 31 Young people ask questions in interviews and questionnaires that researchers do not come up with 2a 1 0