To describe the experiences and perspectives of children and adolescents living with juvenile idiopathic arthritis (JIA).
To describe the experiences and perspectives of children and adolescents living with juvenile idiopathic arthritis (JIA).
We conducted a systematic review of qualitative studies that explored the experiences of children living with JIA. We searched electronic databases (to week 2 of July 2011) and reference lists of relevant articles.
Twenty-seven studies that reported the experiences of more than 542 participants were included. Six major themes were identified: aversion to being different (unrelenting and unpredictable pain, disablement, internal disfigurement, differential treatment, and forced dependency on others); striving for normality (preserving social identity, resourcefulness, sense of community, focus on remission, and mastery over body and pain); stigma and misunderstanding (trivialization of disease, invisible pain, and discrimination); suspension in uncertainty (control versus powerlessness, hope versus disappointment); managing treatment (benefits of taking medicines, respect and involvement in health care, and motivation for physical therapy); and desire for knowledge (medical treatment and advances, lifestyle management).
JIA disrupts a child's sense of normality and impairs his or her capacity for social participation. Children with JIA have a sense of being misunderstood and stigmatized, and they feel perpetually caught between having hope and control over their bodies and overwhelming pain and despair. To increase their confidence, the ability to manage pain, and their resourcefulness for self-management, children need ongoing information about treatments and lifestyle management, strong social support, community advocacy, and active involvement in their own health decision making.
Juvenile idiopathic arthritis (JIA) is the most common chronic rheumatic disease of childhood. It affects approximately 1 in 1,000 children under the age of 16 years (1–3). Children and adolescents with JIA live with chronic or recurrent pain and disability, which can severely limit their ability to complete daily physical tasks and participate in school and social activities (4–6). They can experience delayed growth, joint damage and deformities, pain, swelling, stiffness, and complications including fever, rash, serositis, and vision problems (e.g., uveitis and glaucoma) (1, 6–9). Treatment is often complex and challenging, and involves ongoing medications, monitoring, physical therapy, and surgery in some cases (10, 11).
In children with chronic illness or disability, the characteristics of the condition, functional independence, and psychosocial stress are risk factors for poor adjustment (12, 13). Young people with JIA have to cope with the debilitating impact of arthritis and the demands of treatment while navigating the developmental tasks in childhood and adolescence. Depression, cognitive impairment, and social and behavioral problems have been reported in this population (6, 14–16). To achieve optimal health outcomes, health care services need to be cognizant of patient values, priorities, and experiences. Qualitative research can provide an in-depth understanding into people's experiences that surveys alone may not capture. A single qualitative study may focus on 1 specific topic; however, synthesizing the findings from multiple qualitative studies can provide a wider understanding of children's experiences and perspectives across different topics, settings, and contexts. Thematic synthesis of qualitative studies involves a high-level analytical abstraction of primary findings to develop new conceptual frameworks (17).
A review is needed to identify a range of health care needs of children with JIA. Their experiences and perspectives can inform the development of health care interventions that are relevant and effective for improving health, psychosocial, and treatment outcomes. This study aims to synthesize qualitative studies on the experiences and perspectives of young people living with JIA.
Juvenile idiopathic arthritis has a profound impact on children's lives, which is reflected in the 6 major themes identified in the synthesis: aversion to being different, striving for normality, stigma and misunderstanding, suspension in uncertainty, managing treatment, and desire for knowledge.
Children need ongoing information about treatments and lifestyle management, strong social support, community advocacy, and active involvement in their own health decision making to increase their confidence, ability to manage their pain, and resourcefulness for self-management.
We included qualitative studies published in peer-reviewed journals that explored the experiences of children and adolescents (age <21 years at the time of the study) who had been diagnosed with JIA before age 16 years. Non-English articles were excluded due to the lack of resources for translation. Articles were excluded if they used structured questionnaires and reported only quantitative data. Observational epidemiologic studies, editorials, reviews, nonresearch articles, and studies that did not elicit data from children and/or adolescents with arthritis were also excluded.
JIA is currently the accepted term for chronic inflammatory arthritis of childhood, but this condition has been known by a number of other terminologies in the past. Therefore, search terms for juvenile arthritis, including rheumatoid arthritis, chronic arthritis, idiopathic arthritis, and ankylosing spondylitis, were combined with terms relating to children and adolescents, and then combined with terms and text words for psychological and social concepts, communication, life events, and quality of life. The full search strategy is provided in Supplementary Appendix A (available in the online version of this article at http://onlinelibrary.wiley.com/journal/10.1002/(ISSN)2151-4658). The searches were carried out in Medline (1948 to week 1 of July 2011), Embase (1980 to week 27 of 2011), and PsycINFO (1806 to week 2 of July 2011). We also searched Google Scholar, a reference list of relevant studies and reviews. An author (AT) screened the titles and abstracts and discarded those that were ineligible. Full texts for potentially relevant studies were examined for study eligibility.
We assessed the explicitness of reporting of each primary study. This can provide contextual details for readers to assess the transferability and trustworthiness of the study findings to their setting (18). Also, assessing study reporting ensures a thorough reading and understanding of each article by the reviewers. We used a framework that was developed for the reporting of qualitative studies, which included criteria specific to the research team, study methods, context of the study, analysis, and interpretations (18). The domains are provided in Table 1. Two authors (AT and JJ) independently assessed each study using the framework, and any disagreements were resolved by discussion.
|Experience or training in qualitative research||–||–||–||+||+||–||–||–||–||–||–||–||–||–||–||–||–||–||–||–||–||+||–||+||+||–||–|
|Relationship with participants established prior to study commencement||–||–||+||+||+||–||–||–||–||+||–||–||+||–||–||–||–||–||–||–||–||–||+||+||–||–||–|
|Selection strategy (e.g., snowball, purposive, convenience)||–||–||+||+||+||+||+||+||+||+||+||+||+||+||+||+||+||+||+||+||+||–||+||+||+||+||+|
|Method of approach/ recruitment||–||–||–||–||–||+||+||+||+||+||+||+||+||+||+||+||+||+||+||–||+||+||+||+||–||–||+|
|Number/reasons for nonparticipation||–||–||–||–||–||+||+||–||–||+||+||–||+||+||+||+||+||–||+||–||–||–||–||–||–||–||–|
|Venue of data collection||–||+||+||+||+||+||+||–||+||+||+||+||+||+||+||+||+||+||+||+||–||–||+||–||+||–||–|
|Presence of nonparticipants||–||–||–||–||–||+||–||–||–||+||–||–||–||+||+||–||–||–||+||+||–||–||–||+||–||–||+|
|Description of the sample||+||+||+||+||+||+||+||+||+||+||+||+||+||+||–||+||+||+||+||+||+||+||+||+||+||+||+|
|Questions, prompts, or topic guide||+||–||+||+||+||+||+||–||–||+||+||+||–||–||+||+||+||+||+||+||+||+||+||+||+||–||+|
|Data (or theoretical) saturation||–||+||–||–||–||–||–||–||–||–||–||–||–||–||–||–||–||+||–||+||+||–||+||–||–||–||–|
|Number of data coders||–||+||–||–||–||–||+||+||+||+||–||–||–||+||+||+||–||+||+||–||+||–||+||+||+||–|
|Description of coding tree||–||+||+||+||+||+||+||–||–||+||–||+||–||+||+||+||–||+||–||+||+||+||–||+||+||+||–|
|Derivation of themes/findings||–||+||+||–||–||+||–||+||+||+||+||–||–||+||+||–||+||+||+||+||+||+||+||+||+||+||+|
|Protocol for data preparation and transcription||–||+||+||–||–||+||–||+||+||+||+||–||–||–||+||–||+||+||–||+||+||+||+||+||+||+||+|
|Use of software||–||–||–||–||–||–||–||+||+||–||+||–||–||–||–||–||–||+||+||–||–||–||–||–||+||+||–|
|Participant feedback or member checking on findings||–||–||–||–||–||–||–||–||–||+||–||–||–||–||+||–||–||–||–||–||–||–||–||–||–||–||+|
|Participant quotations or raw data provided (picture, diary entries)||–||+||+||–||–||+||+||+||+||+||+||+||+||+||+||+||+||+||–||+||+||+||+||+||+||+||+|
|Range and depth of insight into participant perspectives (thick description provided)||–||+||–||–||–||+||+||+||+||+||+||–||+||+||+||–||+||+||–||+||+||–||+||+||+||+||+|
We used the technique of thematic synthesis to synthesize the findings (17). Thematic synthesis draws from meta-ethnography and grounded theory. Codes are arranged into descriptive themes and further interpreted to develop analytical themes (16, 19–21). Thematic synthesis is more amenable to synthesizing multiple studies, including “thinner studies,” whereas other methodologic approaches for qualitative syntheses (e.g., meta-ethnography  and critical interpretive synthesis ) are more suitable for synthesizing primary qualitative studies offering richer detailed descriptions. Also, thematic synthesis involves the use of computer software to code the results of the study, which can enhance transparency of the analysis process (24). For each article, we extracted all the participant quotations and text under the “results/findings” or “conclusion/discussion” sections of the article. These were entered verbatim into HyperRESEARCH software (ResearchWare, version 3.0) for coding qualitative data. An author (AT) performed line-by-line coding of the findings of the primary studies, identified descriptive themes, and then developed analytical themes. For each article, the same author coded the text and recorded concepts that focused on the experiences and perspectives of children living with arthritis and identified common and divergent concepts. Subsequent articles were analyzed similarly, and we translated the concepts from one study to another by adding coded text to existing concepts or creating a new concept when necessary. The author reread each article to ensure that all concepts were integrated and the relationships between the key concepts of each study were explored. The preliminary coding framework was discussed among the authors (AT, JJ, and DSG). We examined the concepts for similarities and differences and grouped them into descriptive themes and subthemes. To extend the findings offered by the primary studies, we used Freemind 0.9.0 Beta 14 (Source Forge) to map the interrelationships between themes and to develop higher-order analytical themes.
Our search yielded 1,663 articles. (Figure 1). Of these, 1,582 were excluded based on title and abstract review because they did not meet the inclusion criteria. Of the remaining 81 studies, 54 were excluded because they were quantitative surveys, epidemiologic research or nonresearch articles, or did not include perspectives from children or adolescents with arthritis. Twenty-seven studies involving more than 542 children and adolescents were included (1 study did not report the number of participants). The study characteristics are provided in Table 2. The studies were conducted in the UK, Canada, Australia, The Netherlands, Finland, Sweden, Belgium, and the US. Across all the studies, focus groups, interviews, observations, diaries, and drawings were used to collect data.
|Study||Country||Participants with JA, no.||Age, years||Diagnosis||Data collection||Conceptual methodologic framework||Analysis||Principal participants' experiences explored|
|Baars, 2005 (46)||UK||54||8–16||JIA||Focus group||–||–||Health-related quality of life; develop tool|
|Barlow, 1999 (47)||UK||10||8–15||JCA||Focus group||–||Thematic analysis†||Needs and preferences, and perspectives on psychoeducational interventions|
|Batthish, 2005 (48)||Canada||14||6–18||JIA||Face to face interview||Delphi study‡||Content analysis§||Perceptions of active disease|
|Beales, 1983 (49)||UK||75||7–17||JCA||Face to face interview, drawings||–||–||Physical nature of illness and clinical treatment|
|Beales, 1983 (25)||UK||39||6–17||JCA||Face to face interview||–||–||Meaning of joint sensations and pain|
|Beresford, 2003 (50)||UK||17||10–16||JCA||Face to face interview, group discussion||–||Framework analysis¶||Communication with health care professionals|
|Berry, 1993 (51)||US||54||6–17||JRA||Face to face interview||–||Content analysis||Understanding and conceptualization of illness|
|Britton, 2002 (52)†||UK||9||7–13||JIA||Face to face interview, video diary, diary account||Grounded theory#||Grounded theory analysis||Experience of illness|
|Britton, 2002 (53)†||UK||9||7–13||JIA||Face to face interview, video diary, diary account||Grounded theory||Grounded theory analysis||Experience of exercise and splinting programs|
|De Monte, 2009 (40)||Australia||13||8–16||JIA||Face to face interview||–||Thematic analysis||Experience of living with arthritis and participation in exercise programs|
|Eyckmans, 2011 (54)||Belgium||11||18–30||JIA||Face to face interview||Grounded theory||Grounded theory analysis||Meaning of arthritis|
|Fuchs, 2008 (55)||The Netherlands||1||18||Polyarticular arthritis||Face to face interview||Self-confrontation method**||–||Impact of arthritis on adolescent life|
|Guell, 2007 (56)||UK||4||7–16||JIA||Face to face interview, observation||Ethnography††||–||Daily living and coping|
|Hackett, 2003 (57)||UK||12||7–11||JIA||Face to face interview||Phenomenology‡‡||Thematic analysis||Perception of play and leisure|
|Hutchinson, 2007 (58)||UK||6||16–19||JIA||Face to face interview||Phenomenology||Colaizzi 7-stage process||Patient learning experiences|
|Konkol, 1989 (59)||US||50||2–22||JA (polyarticular, pauciarticular, systemic rheumatic disease)||Open-ended questionnaires, face to face interview||–||Content analysis||Perception and impact of illness|
|Kyngas, 2004 (60)†||Finland||6||13–17||JRA||Face to face interview||Grounded theory||Content analysis||Support network|
|Pollock, 1997 (61)||Canada||1||17||JA||Face to face interview||–||Thematic analysis||Meaning of play|
|Power, 2003 (62)||US||42||6–13||JRA||Video-taped observation of interaction tasks and conversations||–||Content analysis||Interaction between children and mothers|
|Sallfors, 2002 (26)||Sweden||22||6–17||JCA||Face to face interview||Grounded theory||Grounded theory analysis||Life situation and psychosocial processes|
|Sallfors, 2009 (63)||Sweden||6||14–17||JCA||Face to face interview, diaries||Grounded theory||Grounded theory analysis||Daily living|
|Secor-Turner, 2010 (64)||US||9||14–28||JA||Focus group interview||–||Content analysis||Challenges and experiences|
|Shaw, 2004 (33)||UK||30||13–30||JIA||Focus group||Phenomenology||Interpretative phenomenologic analysis||Transitional care|
|Shaw, 2006 (32)||UK||8||14–16||JIA||Focus group||Phenomenology||Interpretative phenomenologic analysis||Prevocational and early employment needs|
|Stinson, 2008 (10)||UK||36||12–20||JIA||Face to face interview, focus group||–||Thematic analysis||Self-management needs|
|Tuchman, 2008 (34)||US||NS||15–21||JRA||Face to face interview||–||–||Transition to adult care|
|Woodgate, 1998 (65)||Canada||4||13–16||JRA||Face to face interview||Grounded theory||Grounded theory analysis||Perspectives of illness|
Comprehensiveness of reporting was variable with studies reporting between 6 and 21 of the possible 26 items (Table 1). Of the 27 studies, 24 provided details on participant selection and 26 described the participant characteristics. Questions, prompts, or a topic guide were provided in 21 studies, and audio/visual recording was reported in 22 studies. Only 7 studies reported the use of software to analyze qualitative data, and 3 reported on whether participants were given the opportunity to provide feedback on preliminary findings. Raw data including quotations, diary entries, or drawings were provided in 23 studies. Nineteen studies were independently deemed by 2 authors (AT and JJ) to provide a range and depth of insight of children or adolescent perspectives.
We identified the following 6 major themes: 1) aversion to being different (unrelenting and unpredictable pain, disablement, internal disfigurement, differential treatment, and forced dependency on others); 2) striving for normality (preserving social identity, resourcefulness, sense of community, focus on remission, and mastery over body and pain); 3) stigma and misunderstanding (trivialization of disease, invisible pain, and discrimination); 4) suspension in uncertainty (control versus powerlessness, hope versus disappointment); 5) managing treatment (benefits of taking medicine, respect and involvement in health care, and motivation for physical therapy); and 6) desire for knowledge (medical treatment and advances, lifestyle management). To illustrate each theme, selected participant quotations and explanations offered by the authors from the included studies are provided in Table 3. Some themes were not mutually exclusive and the conceptual links between themes are depicted in Figure 2.
|Aversion to being different|
|Unrelenting and unpredictable pain||“I've always wondered what it's like to be ‘normal.’ I mean what they feel like, how they move, when they have ‘pain.’ For years I thought everybody was in pain. Then I realized they weren't” (52).|
|“The perfect day would be to get rid of your illness just for a day … I would do anything for that … That would be living … It would be great to know what it's like not to be in pain … I'm in pain all the time” (26).|
|Disablement||“You can't do everything that the other kids do. You are depressed sometimes when your friends are out running and jogging and riding their bikes a lot and stuff and you can't because you have asthma or arthritis or diabetes” (65).|
|Both the pain and the rheumatic disease itself made the children extremely tired, and they often did not manage to live a life like their healthy peers: “It's like being exhausted all the time. Because it's very tiring … always being in pain.” Another dimension of tiredness is pointed out: “And that's what you get so tired from … mentally tired … Not knowing what it is and why” (26).|
|“Well now, I'm about to turn 20. Imagine that I live to be 80 or 90. Well that would mean another 60 to 70 years of that rheumatic pain and that prospect frightens me, you know but, then by that grand age, my personal mobility will be so limited, that I'll probably be incapable of most things” (54).|
|Internal disfigurement||“When my fingers ache, I imagine the bones swelling and getting bigger all the time stretching the skin. It's horrible. It makes me sick” (25).|
|Differential treatment||“Yeah, they treat you different. They treat you like you are a little baby or that you are the nicest person in the world. They would be so nice to you and that's not the way you want to be treated. You want to be treated like a normal human being” (65).|
|Forced dependency on others||The children, except the youngest ones, talked about living in dependency for hours and days and that it was difficult to develop enough self-confidence to dare to say, “No, I do not want your help.” The risk of hurting someone from whom the children would later need help could be important, which was expressed as follows: “It's a bit difficult you know … to say this every time you get to the door, ‘Would you mind opening for me?’” (26).|
|Striving for normality|
|Preserving social identity||Since her arthritis is in remission, Laura never tells people she meets of her disease. She is afraid people will treat her differently if they know about her arthritis… . “What would people think and say about me if they knew … ?” (55).|
|“The thing I would be most scared about right now is, because there are so many diseases in this world, when people hear the word disease, they think it's contagious … so all my friends know that I have this disease and I'm kind of scared to tell other people just in case they don't want to be my friend” (65).|
|Resourcefulness||“Anything I do which makes me more ‘normal’ gives me more confidence, from silly little things like poster? To major things like shopping with my friends without my mum around” (52).|
|Especially important was finding new things to do in order to replace old interests that were no longer possible to carry out because of the illness: “Yeah, I was going to say sometimes you can make the best out of it by finding a new thing” (65).|
|“Obviously I can't be a sportsman because it's going to be hard on my joints, but the jobs that I want to do, is what I'm interested in and I think they are able to be catered to your needs” (40).|
|“ … just plan them ahead more often, accept ‘punishment,’ as they call it, in the form of pain, and develop creative strategies to gain more control over the pain” (56).|
|Some would engage in another activity as a way of distracting themselves, and others would ignore the situation (10).|
|Sense of community||The strong support perceived in the rheumatic support groups makes these settings important in helping the teenage girls to strengthen their own capabilities and recourses. The data show that support groups increase their hopes of managing a life with a chronic disease. Sharing their inner thoughts and similar problems and fears also seems to be a good strategy for strengthening their self-image. Knowing that you are not alone with a chronic and unpredictable disease gives a sense of community: “I have been at an appointment at the clinic together with girls with arthritis, all of them in my age. It makes me happy to meet them, and it is relaxing. We have a lot in common. The disease is invisible for them, too. We can learn from each other. We understand each other without saying a word” (63).|
|Focus on remission||“Eventually you'll have a time slot where it will descend and you will feel better, get on with your life and then it comes back again” (65).|
|Mastery over body and pain||“Arthritis means that the body is out of control, attacking its own tissues. Controller controlling the body, to be more precise is, in fact, one of the key components of the therapy, and through the language of the doctors and therapists the children immediately become acquainted with this” (56).|
|“Pain won't keep me from doing what I want to do. It has already stopped me too much.” Being mentally prepared for a potential negative scenario is a strategy for mastering a body in pain. The illness is often unpredictable. One of the girls said, “I prepare myself almost every day to wake up having excruciating pain” (63).|
|In her diary she describes several situations in which this becomes clear to her, i.e., constantly testing her body by working 60 hours a week to feel in control of her arthritis. “For eight months I worked in a busy restaurant with an outside pavement; it was exhausting but I knew I could do it. I wanted to extend my limits and did!” (55).|
|Stigma and misunderstanding|
|Trivialization of disease||“One of the teachers has got it, mum keeps telling me to tell her it's not the same” or “they were like, my Nan has got arthritis, but obviously it's not the same … I might have to live with it for the rest of my life” (58).|
|Invisible pain||Since the qualities of pain are invisible and unpredictable, people around the girls often distrust their pain. It seems to be hard to gain sympathy for an invisible ailment and misunderstandings appear both in relations to friends and to professionals for which the girls were also mentally prepared (63).|
|When someone's pain was invisible, it seemed to be more difficult for others to understand and for the environment to respond: “The worst thing was that my disease was invisible … because I didn't have a bandage or anything” (26).|
|“People don't understand that one day you can be okay and the other day, that you can't. Some of my friends don't understand that I can't do things as well as they can. I would prefer them [friends] to know a lot about it because then they'd know how I feel and they would understand more … When I have been playing a lot of games and when I sit out, everyone thinks that I'm doing it because I really don't want to do any of the games, but, it's because it's really hurting me and I can't” (47).|
|The reactions from the people around them affected the children considerably, and the children stated that other people distrusted their pain. “My peers thought it was strange that one day you arrive on crutches and the next you can hardly walk … and then you can walk normally … It didn't make sense to them … I feel that I just can't cope with it again … It was so difficult when they wouldn't believe me.” It was important to the children to have support and to be trusted (26).|
|Children wanted others to understand their needs but did not feel able to explain the nature of JCA themselves (47).|
|One youth described the main challenge as “having your friends understand what you're going through” (64).|
|Discrimination||“They'll just see the wheelchair and that's it. Even though I'm clever and get good grades and my CV is going to be quite full because of other things I do, I still think I'll get turned down for someone else who's going to be able to the job all the time, whereas like us, if we have a flare-up, then we won't be able to work” (32).|
|“If they find out you've got a disability you never hear from them again” (32).|
|Suspension in uncertainty|
|Control vs. powerlessness||The fluctuating essence of arthritis seemed a key cause that might impede the child and family from coming to terms with or getting used to living with this illness (52).|
|The children and parents described falling between the world of the “well” and that of the “disabled” because they had periods in both worlds but felt understood by neither (52).|
|“You get so disappointed, because suddenly you're better … and then you're worse again. All the time you're hoping that you'll be better … but sometimes you got worse.” The results indicate that there is variation in pain and, therefore, the children could live like their healthy peers one day, only to realize another day that “the pain had taken over.” They expressed a sense of frustration, powerlessness, and uncertainty about the future (26).|
|“It was dreadful … because I thought I'd die then … I said to Mum … I can't cope … Will it be like this for a whole week? I didn't know what to do” (26).|
|“It's sort of difficult not to know if … today or tomorrow … my joints will be okay. That's what's a bit difficult … never knowing. You have to do it day by day” (26).|
|Hope vs. disappointment||One category, frequently occurring in the interview data, was identified as a core concept and labeled oscillating between hope and despair (26).|
|Benefits of taking medicine||“You get used to having it, you get used to taking your pills and you are used to doing everything that you are supposed to do and exercising it and all that kind of stuff and it goes away. Then you forget all about having it and then if it comes back, you have to get used to taking your pills again” (65).|
|“You take your pills for your own benefit, it's like you are not taking your pill to make your parents feel better, you are taking your pill to make you feel better so if you don't take your pill it's your own fault” (65).|
|Children wanted to know why they were taking particular medications and they wanted to be informed of potential side effects (47).|
|Respect and involvement in health care||“You've really got to ask people like us what we want … otherwise you've just got a load of people who think they know about arthritis. It doesn't work … It's not doing anything for anyone” (47).|
|“Not knowing what all these tests were for was a lot scarier than at least knowing the reasons for them. We are children, not stupid” (56).|
|In regard to privacy, the majority of participants strongly felt that the presence of other health professionals, particularly medical students, during the consultation significantly inhibited communication between themselves and their doctor, even in situations where a good rapport existed between the adolescent and his/her doctor: “You don't tell the doctor anything because you don't want them [student doctors] to hear” (50).|
|Motivation for physical therapy||The majority identified that they were doing exercises to prevent their joints from stiffening and stay moving. The comment made by Lisa (age 12 years), “If you don't move your joints you lose them,” is a sound example of this understanding. Immediate benefits of the exercises were reported; however, the children did not appreciate and understand the long-term benefits (40).|
|Desire for knowledge|
|Medical treatment and advances||“I'd like to know what could happen, so you've got no surprises later on. I'd like to know what I was taking them for [tablets]. Not just take them” (47).|
|In exploring use of other resources, 4 of 6 participants gained information from the internet and some had used multiple resources. LH2 used the local library to get books on arthritis. This led to disappointment as the books did not explain exactly how the drugs worked and some evidence was conflicting, e.g., “Like how folic acid, it doesn't know how it works but it does, methotrexate it doesn't know how it works but it does … I get told another thing by the book, and if anything could be proved it would be that” (LH2) (58).|
|Followup patient education was also highly valued by the participants. The adolescents really needed new information all the time. They wanted to know about the new methods of investigating their condition, new medications, and research results (67).|
|Lifestyle management||Children felt that current interventions failed to explain how JCA may affect school life, friendships, social lives, and family functioning. More specifically, children wanted to know in advance that they may not be able to participate in physical education or go cycling with friends, and they wanted to know that they may be teased or bullied. They wanted to know how they could deal with these situations when they arose. Children were adamant that they should not be shielded from the more negative aspects of JCA and its treatment. As one child said, “I want to know everything” (47).|
|Followup patient education would also be scheduled and planned beforehand. “The problem is that I do not get information any more. I have had arthritis for 5 years. When I got the diagnosis, I had very good patient education, but after that nothing … I am interested in new information concerning arthritis and its treatment but nobody says anything to me. Education should be continuous … .” (67).|
|Seeking information about the illness is another way to control and have influence over a body in pain. The teenage girls in this study sought information on the best ways of managing pain and illness. This resulted in their becoming active educated consumers, empowered through access to information from health care professionals, support groups, magazines, the internet, etc. One teenage girl wrote in her diary, “My doctor explained a lot that I did not know previously and she told me that I really can be healthy again” (63).|
|“I think having a disability is like a financial burden at times. I think maybe if they could give us advice on how to access some financial help to finance our way through college or uni” (32).|
JIA had a profound impact on the participants' sense of normality. The constant and unpredictable pain, disablement, perceived internal disfigurement, being treated differently, and being forced to depend on others made them feel different. They despised feeling different and felt frustrated, fearful, and powerless.
The intense and episodic nature of pain disrupted the participants' sense of normality and their ability to lead lives like their healthy peers. Joint stiffness and pain could be mild one day, but at the next moment be so severe that it would inhibit movement. Pain was perceived to control and restrict the body. During phases of pain, participants felt tired, powerless, and incapacitated.
The joint stiffness and pain imposed severe restrictions on the participants' ability to engage in ordinary physical activities including walking and running. They were frustrated at being unable to engage in sport and social activities with their friends. Many believed they were missing out and felt “sad” and “depressed.” A few were worried about their appearance and awkward movements. The pain was often an unyielding and aggravating reminder of their disability.
Arthritis was described and depicted as a “horrible” (25) disease manifesting in the joint areas. Both verbally and in drawings, some children gave graphic accounts of joint, bone, blood, cartilage, and joint fluid abnormalities occurring within their bodies. The grotesque imagery used to express the perceived physical and internal abnormalities was often expressed with disgust.
Some felt a sense of overprotectiveness from others or perceived they were receiving preferential treatment. However, they wanted to be treated like their normal peers.
A constant need to depend on others for help with ordinary tasks such as opening a door undermined their self-confidence. Some were afraid to try and complete tasks on their own. Yet some participants were also hesitant to reject help from others in order to avoid hurting them, in case they needed their assistance later on.
Striving for normality encompassed attitudes and strategies participants used to cope with the profound sense of being different or incapacitated to achieve their full potential. They did not want to be socially labeled as sick or disabled, pursued alternative social activities they could participate in, accessed social support, focused on remission, and learned to master their bodies and pain.
Some participants were adamant about not being labeled as a sick or disabled person and wanted to avoid sympathy from others. A few believed their peers would think that arthritis was contagious and they were fearful of rejection. For these reasons, many kept their diagnosis a secret from others.
Despite being unable to participate in a range of activities, participants sought alternative activities in which they were able to engage. This would require careful planning involving well-timed pain management therapy before and after an activity. They valued being able to do activities with their peers (e.g., shopping), as this would increase their confidence and sense of normality. In terms of vocational opportunities, participants accepted their limitations and opted for a job or career in which they were interested and that would cater to their needs.
Support from family and other young people with a shared experience was particularly valued by the participants. Interacting with other people with arthritis promoted a sense of normality in their lives and allowed participants to share their experiences, emotions, and coping strategies.
To cope, some participants focused on remission and pain-free moments. During times of pain, they expressed a hopeful anticipation of a time when they would be free of pain and symptoms.
Some participants learned to take control over their bodies and refused to allow pain to limit their abilities and potential. With determination, they pushed themselves beyond their physical limitations to work or play with their friends. A few mentally prepared themselves for the pain they would have to endure during the day.
The participants felt that others did not recognize the seriousness and unpredictability of their arthritis, which was considered by others as a mild disease that only occurred in older people, or was brought on by “cracking your fingers.”
Many participants were deeply frustrated and hurt by the lack of empathy and understanding from others who could not see the “invisible” (26) arthritic pain and symptoms. This was even described as the “worst” thing about living with arthritis. Given the fluctuating intensity of pain, some children were accused of feigning pain to excuse themselves from various school activities. Some struggled to explain arthritis and its impact and were reluctant to talk about the nature of their illness.
Among the older adolescents, some felt their vocational and career opportunities could be limited. They believed potential employers may discriminate against them due to their physical disabilities
Because of the unpredictable nature of pain, the participants believed they were trapped in a perpetual limbo, oscillating between having a sense of control over their bodies and losing all control as moments of intense pain overwhelmed them.
Similarly, the participants contended with undulating emotions between the extremes of hope and despair. When experiencing pain and sickness, they felt depressed, defeated, and at moments wanted to give up. At other times, usually when the pain and symptoms were mild or imperceptible, they were hopeful and believed in remission and recovery.
Some participants believed that taking medicine was important for their own health and were able to integrate it into their daily routine. Other children found it difficult to take their medicines and were apprehensive about blood tests as this would potentially mean an increase in their dose of medication. Some were aware of the potential dependency of pain medication and were cautious about taking pain medications too frequently.
The participants wanted active participation in clinical decision making. They felt health care providers should explain the various tests and procedures as this would offer a sense of reassurance. Familiarity, a good rapport, and a trusting relationship with health care professionals were highly valued. To the participants, it was important that they could access support from health care providers who understood their illness, pain, and emotional experiences.
Most participants believed that physical exercises were necessary for keeping their joints mobile. They were willing to participate in exercises if they were perceived to be fun. However, some had other priorities including school work, or they were too tired, busy, or bored from doing repetitive physical exercises.
The participants wanted to know how the medications worked and their potential side effects. They were also keen to know about medical advances and research findings as this offered a sense of hope.
Knowledge about the impact of living with arthritis on school, relationships, family functioning, finances, and daily living was important to the participants. Specifically, they wanted to know about the activities in which they were not allowed to participate, and that they may be rejected or bullied by their peers. When equipped with practical information, the participants felt empowered to manage their pain. The knowledge about the possibility of remission and recovery gave some participants a sense of hope.
JIA profoundly disrupted the children's sense of normality. Recurrent and intense pain restricted their ability to complete daily physical tasks and served as a frustrating reminder of their disability. Their social and school participation was limited by pain. They felt perpetually caught between having hope and control over their bodies, and overwhelming pain and despair. Children and adolescents with chronic arthritis also had a sense of being misunderstood and stigmatized, and struggled to preserve their social identity by keeping their illness a secret. To cope with the debilitating impact of arthritis, they focused on remission and recovery, took initiative to seek alternative activities or career pathways that could accommodate their physical limitations, gained information about the illness, treatment, and self-management, and accessed social support. They learned self-management and implemented strategies to master their body and pain.
In this study, we used software to code the data and record an auditable development of themes that can be used to assess whether these findings reflect the original findings in the primary studies. By making comparisons across studies, we developed a new conceptual framework (Figure 2) that illustrates relationships between themes (which are not apparent in individual articles) to provide a more complete and holistic understanding of the spectrum of the impact JIA can have on children and adolescents, as well as strategies that may facilitate positive adjustment and coping. Some studies involved a small number of participants; however, qualitative research usually selects information-rich participants and does not seek to attain statistical representativeness and generalizability. A potential limitation of the study is that we did not assess the degree to which the research findings informed theory development, which is typically conducted in a critical interpretive synthesis (19). Instead, in accordance with thematic synthesis methodology (27, 28), we conducted a quality appraisal of criteria relating to the reporting of procedural aspects of the research, to assess reliability, validity, and appropriateness of methods for ensuring that the findings are grounded in participants' perspectives.
Our findings enhance existing theoretical models (13) by providing more clarity about children's perspectives in the context of JIA. For example, in our study, the theme disablement related to multiple dimensions of physical disability, psychosocial stress (e.g., frustration), and self- perception. The unpredictable pain meant they felt suspended in uncertainty, vacillating between control and powerlessness, and hope and disappointment. Arthritis is an invisible disease, often perceived to occur only in older adults. Therefore, children felt misunderstood and stigmatized and strived to preserve their social identity.
In addition to psychosocial issues, our findings encompass children's experiences and perspectives of medical management and health services. They suggested that health care providers should deliver holistic care, including emotional support and lifestyle management, and give adequate information about tests and procedures until children felt a sense of reassurance. Although physical therapy was believed to be important, children suggested exercises needed to be enjoyable and integrated among their other priorities, including school commitments. They also wanted to be informed about medical research and advances in JIA to gain a sense of optimism for the future. Children need ongoing access to psychosocial and educational support to promote positive adjustment and coping. Children and adolescents wanted ongoing information about their illness, medical side effects, how the treatment works, and current information on medical advances and research into chronic arthritis. They were keen to learn strategies on how to manage their pain and lifestyle to counteract the profound impact arthritic pain exerted on their mental, emotional, and physical capacities. These aspects of support and education might be best incorporated as part of a multidisciplinary team care plan providing age-appropriate education for patients and families through the course of the disease.
It is estimated that 50% of children will continue to have chronic arthritis into adulthood. Poor self-perceived health state, emotional problems, social isolation, and high rates of unemployment have been reported in adults diagnosed with arthritis in childhood (29). This has important implications for transition, defined as “a purposeful, planned movement of adolescents and young adults with chronic physical and medical conditions from child-centered to adult oriented health-care systems” (30). Transitional care in rheumatology is widely advocated and has been shown to improve health-related quality of life (29, 31). In our review, only 3 qualitative studies have elicited views from adolescents on transition (32–34). Future work is needed that actively involves patients during the whole process of transition and to ensure that perspectives, priorities, and concerns are addressed in order to improve medical and quality of life outcomes in the long term.
Camps or social outings can facilitate learning of coping skills, as well as promote a sense of normality and social inclusion. Recently, internet-based initiatives for information provision, networking, and self-management in juvenile arthritis have been developed with promising results for feasibility, user satisfaction, and behavioral and health outcomes (35–37).
While physical therapy may have potentially short- and long-term benefits (38–40), exercise programs could incorporate enjoyable activities, incentives, a range of different tasks, and goal setting, which may promote motivation for adherence to physical therapy.
Being misunderstood and discriminated against, especially in school, intensified children's feelings of stress and grief. Even in adulthood, studies found that adults with JIA encountered discrimination in their workplace and had high rates of unemployment (41). Therefore, we recommend that advocacy in schools and workplaces should be an integral component of the health care service for children and young people with arthritis. A health care professional may liaise with the school or workplace to promote awareness and ensure that the individual's needs are supported. Programs to promote successful transition from to school to work are also recommended.
Although a number of general pediatric quality of life instruments are available and have been used in pediatric rheumatology (3), the themes identified in this review may inform the development of specific quality of life instruments for young people with JIA. The Juvenile Arthritis Quality of Life Questionnaire (42, 43) addresses psychosocial function based only on the frequency of problematic behaviors and emotions as observed by older children or their parents. Our review identified additional concepts relating to the child's self-perception that provide insight into the severity and meaning of the impact of juvenile arthritis on patients that current instruments do not assess. Further qualitative research is also recommended to elicit their experiences and perspectives on issues that have not been addressed, including concerns about the impact of JIA and immunosuppression on fertility.
A meta-systems approach to evidence-based practice for children has been proposed, which acknowledges the importance of a broad range of service sectors including school-based interventions and specialty mental health services (44). Studies also suggest that adjuvant psychological interventions appear efficacious for improving clinical, knowledge, behavior, and psychological outcomes; however, current evidence is of suboptimal quality (45). High-quality research is needed on the effectiveness of mental health interventions addressing the range of emotional challenges and experiences identified in our review, including feelings of being misunderstood and stigmatized, internal disfigurement, being focused to depend on others, and contending with unpredictable phases of pain and physical disablement.
JIA can have a debilitating impact on children and adolescents. Patients must contend with unpredictable phases of incapacitating pain, stigmatization, and physical limitations. Continued information, strong social support, community advocacy, and active involvement in their own health decision making are needed to enhance confidence, the ability to manage their pain, and resourcefulness in children and adolescents with JIA. This may lead to improved treatment and health outcomes among young people diagnosed with JIA.
All authors were involved in drafting the article or revising it critically for important intellectual content, and all authors approved the final version to be submitted for publication. Dr. Tong had full access to all of the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis.
Study conception and design. Tong, Craig, Singh-Grewal.
Acquisition of data. Tong, Jones.
Analysis and interpretation of data. Tong, Craig.