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Daily life participation in childhood chronic disease: a qualitative study
  1. Merel M Nap-van der Vlist1,
  2. Marijke C Kars2,
  3. Emma E Berkelbach van der Sprenkel1,
  4. Linde N Nijhof1,
  5. Martha A Grootenhuis3,
  6. Stefan M van Geelen4,
  7. Cornelis K van der Ent5,
  8. Joost F Swart6,
  9. Annet van Royen-Kerkhof6,
  10. Martine van Grotel7,
  11. Elise M van de Putte1,
  12. Sanne L Nijhof1
  1. 1 Paediatrics, Wilhelmina Children's Hospital, University Medical Center Utrecht, Utrecht, The Netherlands
  2. 2 Julius Center for Health Sciences and Primary Care, Utrecht, The Netherlands
  3. 3 Psycho-oncology, Princess Máxima Center for Pediatric Oncology, Utrecht, The Netherlands
  4. 4 Education Centre, University Medical Center Utrecht, Utrecht, The Netherlands
  5. 5 Cystic Fibrosis Center and Department of Pediatric Respiratory Medicine, University Medical Center Utrecht, Utrecht, The Netherlands
  6. 6 Paediatric Rheumatology, Wilhelmina Children's Hospital, University Medical Center Utrecht, Utrecht, The Netherlands
  7. 7 Oncology, Princess Máxima Center for Pediatric Oncology, Utrecht, The Netherlands
  1. Correspondence to Merel M Nap-van der Vlist, Paediatrics, Wilhelmina Children's Hospital, University Medical Center Utrecht, Utrecht 3508AB, The Netherlands; m.m.vandervlist-3{at}umcutrecht.nl

Abstract

Objective Opportunities to participate in daily life have improved considerably for children with chronic disease. Nevertheless, they still face challenges associated with their ever-present illness affecting every aspect of their lives. To best help these children, we aimed to assess the child’s own perspective on participation and the main considerations that affect participation in a stable phase of disease.

Methods Qualitative study design was applied. Semistructured, indepth interviews were conducted and analysed by a general inductive approach using constant comparison, coding and categorisation. Children 8–18 years old with a chronic disease were recruited from a cohort study involving cystic fibrosis, autoimmune disease and post-treatment paediatric cancer.

Results 31 of the 56 (55%) invited patients participated. From the perspective of children with chronic disease, participation is considered more than merely engaging in activities; rather, they view having a sense of belonging, the ability to affect social interactions and the capacity to keep up with peers as key elements of full participation. Some children typically placed a higher priority on participation, whereas other children typically placed a higher priority on their current and/or future needs, both weighing the costs and benefits of their choices and using disclosure as a strategy.

Conclusions Enabling full participation from the child’s perspective will help realise patient-centred care, ultimately helping children self-manage their participation. Caregivers can stimulate this participation by evaluating with children how to achieve a sense of belonging, active involvement and a role within a peer group. This requires active collaboration between children, healthcare providers and caregivers.

  • participation
  • chronic disease
  • children
  • qualitative research

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What is already known on this topic?

  • Participation in daily life is crucial for a child’s development.

  • Most adults who grew up with a chronic disease achieved participation milestones later.

  • In order to stimulate participation in children with chronic disease, understanding the child’s perspective on participation is essential.

What this study adds?

  • Children with a chronic disease define full participation as more than engaging in activities.

  • They view full participation as having a sense of belonging, the ability to influence their social interactions and the capacity to keep up with their peers.

  • Children carefully weigh participation against their current and/or future needs, considering the costs and benefits of their choices and using disclosure as a strategy.

Introduction

Participation in daily life plays an important role in a child’s development and helps facilitate a healthy transition into adulthood.1 Participation was defined by the WHO as ‘involvement in a life situation’, such as engaging in social interactions or taking on a role in sports or academia.2 This participation is vital to a child’s well-being; a lack of participation can place a burden on both children and their family, with significant societal costs.3 Although the opportunities to participate have increased considerably in recent decades for children with chronic diseases, they still face challenges associated with an ever-present illness that affects nearly every aspect of their lives, including their ability and/or readiness to participate.4–8

Understanding a child’s own perspective on participation is essential in patient-centred care. Patients with disabilities or asthma have reported that participation is more than attending activities.9 10 Young adults who grew up with a chronic disease often achieved participation milestones at a later age compared with peers and needed extra social support.8 11–13 To stimulate full participation in children with chronic diseases, it is important to understand their perspective, in order to empower and support them in what they perceive as participation.8 14

The aims of this study were to characterise participation in daily life in children with chronic disease and to identify the considerations that affect participation from their perspective.

Methods

We applied an explorative qualitative interview study design. A general inductive approach and the Qualitative Analysis Guide of Leuven method proposed by Dierckx de Casterlé et al were used to describe participation from the children’s perspective.15 16

In line with qualitative sampling strategies, patients were purposefully recruited from a larger cohort of children with cystic fibrosis (CF), an autoimmune disease or patients postcancer treatment at the Wilhelmina Children’s Hospital and the Princess Máxima Center for Pediatric Oncology in the Netherlands.16 Children postcancer treatment were included within 1 year after completing treatment. Children 8–18 years old were eligible if their treating physician determined they were in a stable phase of their disease and able to talk about participation. Maximum variation was sought in age, sex, school absence, and fatigue and pain levels.16 Fatigue was assessed using the general fatigue scale of the Paediatric Quality of Life Inventory Multidimensional Fatigue Scale, which is internationally validated and has good internal consistency (Cronbach’s alpha 0.73).17 School absence was reported over the previous 6 months. Average pain over the previous week was reported on a Visual Analogue Scale from 0 to 10, as this is easily applicable and has higher reproducibility than other pain measurements.18

Children and their parents were informed via the child’s treating physician, received an information letter regarding the (interview) procedures and were given time to decide whether they were willing to participate. After 2 weeks, the researcher contacted them by phone, and after oral consent was given an indepth semistructured interview lasting 60–90 min was planned in the hospital or at their home, depending on their preference.19 Signed informed consent and/or assent were provided by parents and children at the time of the interview.20 The interviewers were trained in qualitative research methods: active listening and asking open-ended follow-up questions to identify the children’s perspectives. If possible, children were interviewed alone, to reduce socially desirable answers. An interview guide based on literature and the research team’s expertise was used (see online supplementary table S1). The interview started with an open-ended question: ‘To what extent does your disease affect your daily life?’ To help younger children express themselves, a card-based tool was developed by a graphic designer in cooperation with the research team. The tool consisted of illustrations showing activities, people in their environment, emotions and contradicting pairs of statements. The cards were based on the interview guide. All interviews were audio-recorded and transcribed verbatim.

Data were analysed using two intertwined strategies: coding and thinking theoretically.20 Data collection and analysis were alternated.15 21 22 Patients were recruited until saturation was reached. Four researchers (MMN-vdV, EEBvdS, SLN and MCK) were involved in the entire process from initial coding towards the final results. They were part of a team of eight researchers from various backgrounds (nursing, philosophy, paediatrics and psychology) who checked findings against transcripts and validated the results. The goal of this researcher triangulation is to repeatedly reassess the data, accurately capturing the content of the qualitative interviews. The first transcripts were read by the complete team and initial concepts were identified. Subsequently, two researchers (MMN-vdV and EEBvdS) started with open coding and discussed findings with a core team (MCK and SLN) to reach consensus. Coding was supported by MAXQDA.23 Constant comparison was established by discussing differences, commonalities and possible explanations for the variation encountered between cases, aimed at a thorough understanding of participation in daily life.15 21

Results

Of the 56 invited patients, 31 (55%) agreed to take part. Included were 11 patients with CF, 11 patients with an autoimmune disease and 9 patients postcancer treatment (table 1). One child was interviewed with her parent present. Reasons for not taking part were already being involved in other studies or finding it too burdensome to talk about their disease. The results are illustrated by quotes of individuals with different illnesses.

Table 1

Patient characteristics (N=31)

Daily life participation

Regarding participation in daily life, children described a large variety of activities within the following domains: real-life participation with peers (eg, school, sports), online participation with peers (eg, online gaming, social media), participation with family members (eg, eating together, going on holidays) and participation with a wider social environment (eg, communities, jobs/internships). Participation was mostly face to face; online participation played an important role among older children.

“Most of the time I talk to other people via internet while I play on the PlayStation… so you do not have to make an effort to go to other people’s house and you can play from your own house.” —Boy with CF, 15 years old, describing the importance of online participation.

Although all participants were in a stable phase of their disease, their perception of being able to achieve full participation varied widely. Some children reported they generally felt able to fully participate, regardless of their limitations, while others reported feeling unable to participate fully due to disease-related limitations. Depending on the situation, children sometimes felt unable to fully participate because they were unable to go out with friends, but were able to fully participate when they were able to overcome this limitation (eg, by having friends come to their house). These relatively large differences between children and situations indicate that from a child’s perspective, the notion of ‘participation’ is more than merely being able to engage in certain activities.

“Even though sometimes you are able to do less, there are always things you can do, so you can do something fun with friends. You do not have to go play soccer, you can also ask friends to go do something at somebody’s home…Then they adapt: we do not have to do this, we can also do something else.” —Boy with juvenile idiopathic arthritis (JIA), 15 years old, describing feeling unable to participate, but being able to fully participate when he is able to adjust the situation to his needs.

From our interviews, we learnt that children who fully participate experienced this as having a sense of belonging, the ability to affect social interactions and the capacity to keep up with peers (table 2).

Table 2

Aspects of full participation and example quotes by children with a chronic disease

Full participation

Having a sense of belonging was identified when a child’s presence was undisputed and self-evident, both to themselves and reflected by their social environment. Children reported they felt invited and/or actively involved, and their social environment adapted naturally to their abilities. This sense of belonging meant that children did not need to disclose their ‘being different’ to others, and their patterns in participation were unaffected by disease-related limitations. In brief, children described this as being different, but not feeling different.

“When I bike slower than others - that they do not ask me to hurry up, but that they adjust to my pace.” —Girl with JIA, 17 years old, describing being actively involved, with her social environment adapting to her abilities, without the need for explicit disclosure.

The ability of children to influence their social interactions was identified as their ability to guide their participation, as social interactions were perceived an important aspect of participation. Children feel they can influence the social process and experience being viewed as an equal partner within these social interactions (eg, they are allowed to participate in a project from home rather than being present); children felt they could disclose their limitations and their environment would adapt to their needs (eg, the child can still design a project, even though they cannot execute it). Children described this as being able to participate in their own way.

“Most of the time I am ordering them to do this and that, because I cannot do all those heavy tasks… So everyone comes to me to ask, ‘What can I do today?’ And I say, ‘Well, this needs to be done or that needs to be done.’ It feels nice that I can arrange something from home… Then I do feel part of it.” —Boy with CF, 18 years old, explaining being an equal partner within his social interactions and participating in his own way.

The capacity to keep up with peers was expressed primarily by adolescents. When they compared themselves with peers, they felt they had a similar level of knowledge, skills or performance. Some children felt able to participate only when they were kept up-to-date regarding social processes (eg, being kept in the loop about what happened in classes they missed). Other children felt they needed specific abilities (eg, the need to run fast enough in order to join a field hockey game) or performance levels (eg, if they were unable to play a computer game for a long time, they no longer had sufficient confidence to play that game with friends).

“Well, in the beginning it was very easy [to play a computer game], but now people have become so much better at it that it… Yes, if you see certain things that people do in the game, you think, ‘How do they do that?’ And a lot of my friends are pretty good too, but I played less. And what I notice is that I do not play with random strangers on the internet anymore, because I’m afraid that they will get angry because I am not good enough.” —Boy post-cancer treatment, 18 years old, describing the need to perform on the same level as peers in order to feel like he can fully participate.

Barriers

In each child, we identified a desire to achieve full participation, but they experienced intrinsic and extrinsic barriers. Intrinsic barriers were related to the children themselves and disease-related factors (eg, fatigue, pain, physical changes, the demands of the treatment regimen, feeling ‘down’ or unable to fit in).

“When I go out for dinner with friends, I leave early because I cannot keep going. I have so much pain that I am not enjoying myself anymore. Then I feel like it’s better for me to go home, than sit there feeling miserable.” —Girl with JIA, 17 years old, describing physical barriers to achieve full participation.

Extrinsic barriers were related to external factors that make the child feel excluded, such as peers not inviting the child, or when a child has the feeling that he/she would not be able to keep up, for example, during sports. These barriers are summarised in table 3, with illustrative quotes in table 4.

“To be too tired to go to the movies or a concert, or to go swimming. And after a while they stop asking you to join. That is stupid.” —Boy with common variable immunodeficiency, 14 years old, explaining feeling excluded due to disease-related limitations.

Table 3

Barriers to full participation among children with a chronic disease

Table 4

Quotes illustrating the barriers to full participation in children with a chronic disease

Considerations

Also, considerations regarding participation were described, defined as weighing costs and benefits. First, children described whether they considered participation desirable in terms of relational and/or personal fulfilment, described as personal pleasure, the belief that participation is beneficial or the knowledge that participation can facilitate accomplishments.

“Compared to my classmates I am in average shape, because some of them do not work out and I do. However, a few bad days can make me have to start all over again. I work very hard to be at the level where I am, but a small flu can throw all my energy away, all the training, just like that. And then I can start all over again.” —Boy with CF, 11 years old, describing sports participation as desirable for health benefits and the ability for future participation.

Second, children considered the feasibility of participation, which included the availability of others, the proximity of the activity, the likelihood that others will allow them to participate and the likelihood that they would not let others down.

“I would not feel comfortable going back to playing field hockey, because I cannot keep up with the others. My physiotherapist says I can do everything, but I know I cannot.” —Girl with CF, 16 years old, describing participation as not feasible, because she feels she will let others down if she participates.

Third, children described the role of barriers. In some situations, children allowed barriers to keep them from participating. A temporary refrain from participation was often justified as a strategy to increase future participation.

“For example with soccer: I join for 15 min, then I wait for 10 min and then I join again for 15 min and so on. If I notice I cannot do it anymore, then I go home earlier.” —Boy postcancer treatment, 12 years old, explaining that fatigue restricts his current participation.

In other situations, children placed a higher priority on participation than on their barriers, investing physical and mental resources. Children used perseverance and tenacity to continue participating despite barriers.

“At first I did not go dancing or drinking alcohol, but now I told my parents and the doctor that I am still a child. I do not like that I am in pain, but I do want to enjoy things and in ten years I cannot go back to redo this. So, then I just grit my teeth and make memories. I prefer that. It is setting priorities.” —Girl with JIA, 16 years old, describing prioritising participation over her physical symptoms.

Finally, some children revealed that the expectation of physical and/or emotional consequences, such as an increase in symptoms, caused them to refrain from participating. Thus, some children typically placed a higher priority on participation, whereas other children typically placed a higher priority on their current and/or future needs, both weighing the costs and benefits of their choices.

“Sometimes I think about it and I know that if I do something, I will have less energy tonight or tomorrow. And then I decide not to do it.” —Girl postcancer treatment, 16 years old, describing that the anticipation of fatigue keeps her from participating.

Disclosure

We define ‘disclosure’ as telling or showing others minor or major aspects of their disease or limitations, making the disease more visible and making it possible for others to adapt accordingly. Children varied in how much they disclosed in different situations. ‘Standing out’ in a negative way while striving to be normal was described as a potentially harmful effect of disclosure for their sense of belonging.

“I do not like being different than others. Therefore, I do not tell them when I do not feel well. I do not need to be special.” —Girl with JIA, 17 years old, describing that disclosure may affect her sense of belonging.

They described a helpful effect as ‘leading to a better understanding among peers’, which allowed their environment to adapt. Not disclosing can be detrimental to achieving participation if the child is unable to keep up. Because the line between helpful and harmful disclosure was described as thin, disclosure is an ongoing consideration for most children.

“If you let people know, they can help you think of solutions. Because otherwise you only have one brain and then it is much harder. Others can be way more creative than you are. When I think it may be best to just not participate, and tell them, they most of the time try to find a solution.” —Girl with systemic sclerosis, 11 years old, describing more understanding and better adaptation by peers as a positive effect of disclosure.

Children who expressed substantial difficulty in participation with peers tended to engage more in family participation. These children noted that the adaptation needed by peers was a barrier too high to overcome. Within their family, acceptance and adaptation occurred naturally. For example, some children reported usually sharing their free time with their parents, whereas other children spent their free time with peers.

“It also had an impact at home, because there I could really be who I wanted to be. When I came home from a long day, my mom was the one noticing that it did not go well.” —Girl with CF, 16 years old, describing that it is easier to be herself, including disease-related limitations, within her family environment.

Discussion

Children with a chronic disease view full participation as more than engaging in activities; rather, they view having a sense of belonging, the ability to affect social interactions and the capacity to keep up with peers as key elements. Children weigh participation against their current and future needs, using disclosure as a strategy. Understanding the child’s perspective will assist clinicians in empowering children with chronic diseases to fully participate.

Most studies base participation primarily on performance or attendance.24 25 Consistent with studies in children with asthma or disabilities, children with chronic disease did not consider full participation to mean participating in the same activities as peers.9 10 26 Other qualitative studies in children with chronic diseases describe similar barriers to participation, such as pain or the unpredictability of the disease.27 28 The described ‘ability to keep up with peers’ is in line with the perceived need to be kept in the information loop by healthy individuals.29 Also, the ability to influence social interactions and the need for their environment to facilitate participation have been described before and have similarities with the perceived responsiveness research.11 30 31 The elements of full participation in our study could be similar in healthy individuals, but achieving autonomy and self-management may be easier for healthy children, since children with chronic diseases are additionally burdened by disease-related limitations. They generally spend more time alone and at home, and report achieving participation milestones at a later age.8 32 Stimulating participation may help them grow into autonomous adults.

One way to stimulate participation for children with chronic disease may be to help them cope with and adjust to their disease and manage their growing state of independence, for example balancing disclosure and striving for normalcy, using cognitive behavioural techniques and positive thinking.12 28 33–40 Healthcare providers can support the child’s pursuit of full participation by working towards a sense of belonging for the child, active involvement and the ability to influence their role among peers. This approach requires active collaboration between child, healthcare providers and caregivers, focusing on the child and their environment.14

A major strength of this study is the focus on the perspective of children with a variety of severe diseases; this is particularly important, as the child’s perspective is a key component in patient-centred care, and with increasing age participation becomes the child’s own responsibility. Children were encouraged to express their thoughts using a card tool. Finally, this study was conducted using methods that optimised validity and reliability, including analyses from data towards theory by a research team with various professional backgrounds.16 22

A limitation is that our results may not necessarily reflect children for whom participation is relatively difficult, as one reason for not taking part was finding it too burdensome to talk about their disease. Second, gatekeeping by healthcare professionals may have prevented some children from being invited.41 Nevertheless, our study revealed sufficient variation in all disease groups with respect to the concepts described. Saturation was reached regarding the features of full participation, although some aspects of the barriers and considerations may not have been fully revealed. As we interviewed Caucasian children only, our results may not fully reflect children from other ethnic backgrounds.

Some aspects that influence participation may not be described by children themselves, such as parental influence on participation or the children’s mental state or personality traits.11 13 31 In future studies, the role of parents and/or healthcare providers in facilitating participation is of interest, as well as approaches that help children overcome barriers to participation.

Conclusions

From the perspective of children with chronic diseases, full participation is characterised as having a sense of belonging, the ability to affect social interactions and the capacity to keep up with peers, rather than engaging in activities. Focusing on full participation from the child’s perspective will facilitate patient-centred care by helping the child self-manage their participation.

Acknowledgments

The authors would like to acknowledge the contribution of the patients and parents who participated in this study, as well as the involved caregivers who included patients and discussed the outcomes of the questionnaires with them. The authors also thank Dr CF Barrett for his assistance with English language editing.

References

Footnotes

  • Contributors SLN and MCK designed the study and were involved in the entire process from initial coding towards the final results, and reviewed and revised the manuscript. MMN-vdV designed the study, collected the data, was involved in the entire process from initial coding towards the final results, and drafted and revised the manuscript. EEBvdS collected the data, was involved in the entire process from initial coding towards the final results, and reviewed and revised the manuscript. LNN designed the study and collected the data, acted as peer reviewer by checking the findings against transcripts and as such validated the results, and reviewed and revised the manuscript. MAG, EMvdP and SMvG acted as peer reviewer by checking the findings against transcripts and as such validated the results, and reviewed and revised the manuscript. MvG, JFS, AvR-K and CKvdE critically reviewed the data analyses and reviewed the manuscript for important intellectual content. All authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work.

  • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

  • Competing interests None declared.

  • Patient consent for publication Not required.

  • Ethics approval The Research Ethics Committee classified this study as exempt according to the Medical Research Involving Human Subjects Act (16–797/C).

  • Provenance and peer review Not commissioned; externally peer reviewed.

  • Data availability statement Data are available upon reasonable request.

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