A qualitative study exploring experiences of treatment adherence in Paediatric Rheumatology - Parents’, Carers’, Children’s, and Young People’s Perspectives

doi:10.21203/rs.3.rs-4358169/v1

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Research Article

A qualitative study exploring experiences of treatment adherence in Paediatric Rheumatology - Parents’, Carers’, Children’s, and Young People’s Perspectives

https://doi.org/10.21203/rs.3.rs-4358169/v1

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Background

There is limited literature in paediatric rheumatology describing holistic lived experiences of children and young people (CYP) and their parents or carers (PC).Improving our understanding in this area is important as they could have implications including treatment adherence. This study aimed to explore treatment experiences of CYP and PC in a paediatric rheumatology service.

Methods

Participants attending day-case ward for various intravenous infusions at a tertiary paediatric rheumatology centre were recruited. Qualitative semi-structured interviews with CYP and PC, were used to collect data. Data were transcribed, quality checked and thematically analysed using NVivo 12.4 to identify findings.

Results

Thirty-two participants (15 CYP between the ages of 6 and 16 years, 17 PC).) took part in interviews lasting 41 minutes and 43 seconds on average. Participants most commonly described experiences using infliximab, followed by tocilizumab and abatacept. Participants described their disease as a wave, oscillating between symptoms and treatment. Experiences of treatments were temporary, which eventually and inevitably failed. Treatment adherence behaviours were influenced through somatic factors (pain, function) and social factors (advice from health professionals, encouragement from friends, family and teachers, practicality of using treatment in relation to school, work and finance) and cognitive factors (fear of needles and fear of specific medication).

Conclusions

Collectively, findings demonstrate experiences of treatment reflect the nature of many paediatric rheumatology conditions oscillating between periods of time when their condition is well-managed, and they are symptom-free and treatment-failure with symptom return. A limitation of the study is interviews were conducted jointly with patients and carers, which may have influenced what participants were willing to say in front of one another. Further work should seek to identify somatic, social and cognitive targets for interventions to improve experiences of treatment in everyday life.

Qualitative research

treatment adherence

patient lived experiences

medicines use

children and young people

carer experiences

behavioural science

Despite being relatively rare, several inflammatory rheumatic diseases are more prevalent in the UK than others. Juvenile Idiopathic Arthritis (JIA) has an age-standardised incidence and prevalence of 5.61 and 43.5 per 100,000 individuals respectively [1]. The incidence of Juvenile-onset Systemic Lupus Erythematosus (jSLE) is between 0.36 and 0.46/100,000[2]. Juvenile dermatomyositis (JD) is a rare muscle disorder that leads to weakness and skin rashes and affects 3 children/1,000,000 yearly [3, 4]. This evidence shows paediatric rheumatologic diseases, although rare when considered as individual conditions, collectively represent a significant population of children in the UK. Although there are pathophysiological difference between rheumatic diseases, the social and cognitive experiences of the diagnostic pathways and treatment services are similar [5]. Recent work in the Lancet, identified psychological or cognitive issues were a research priority for patients with jSLE, JD and JIA, as well as the health professionals looking after them. [6]. There is an opportunity then to consider the experiences of a significant population of children and young people, by examining experiences of paediatric rheumatology services generally, rather than through a disease-specific approach.

Many of the conditions have similar symptoms and treatments, yet, studies investigating patient and carer experiences are focused on one condition or symptom [7]. It is notable that there are different experiences between some diseases, for example, Canadian research has shown children with JIA spends an average of $1,686 more than other children on medication bills, regular visits to doctors, and diagnostic tests.[7] Additional work from Europe has shown the social and economic cost (such as employment and educational absences, as well as travel to and from appointments, in addition to the cost of the actual medication) is high for both patients and carers [8]. The study also revealed the number of joints affected was associated with higher costs, implying that a worse disease state leads to more experiences of economic hardship [9]. Although this does suggest differences in experiences across rheumatic diseases, inflammatory diseases do share a similar pattern of disease progression, involving a series of relapses and remissions over time [10]. However, there is limited research exploring experiences of treatment holistically in paediatric rheumatic diseases across which could be used to develop interventions to support treatment, at scale.

Medical treatments are available in a range of different pharmaceutical formulations, such as solid oral dosage forms like tablets and capsules, liquids and injectable medicines. Although newer oral biologics, such as Tofacitinib,[11] are now available and becoming popular, the majority of treatments are injectable [12]. Previous studies have reported injectable medications are linked to higher treatment anxiety, with ‘needle phobia’ being a key component of reduced treatment adherence [13, 14]. In addition, parents with younger children experience more distress in comparison to older children [14]. In a paediatric endocrinology study, parental and carer treatment anxiety was linked to poorer health outcomes of the children they cared for [15]. All of this means that injectable treatments increase distress, reduce adherence and lead to poor health outcomes. This could be the case in paediatric rheumatology as the mainstay of treatments are injectables. Further work is therefore needed to understand patient, parent and carers experiences of treatment.

Treatment adherence can be explained using the common-sense model of self-regulation (CSM), which describes health behaviours [16, 17]. The model assumes people actively and continuously go through problem-solving when faced with a health issue using ‘common sense’. For example, patients are given a stimulus which represents illness, referred to as a ‘health threat’ or ‘illness representation’ (e.g., a symptom, a blood test reading or other information). Experiences of the health threat can lead to the performance of coping behaviours to manage it. This could include behaviours such as eating a new type of food, trying a new exercise, visiting a health professional or initiating a treatment. These behaviours are then appraised, using a combination of prior personal experiences and knowledge, current symptoms and experiences, as well as social, cognitive and cultural experiences. Following appraisal, these behaviours or ‘coping procedures’ can be repeated if they successfully managed the health threat, adapted if they partially managed the health threat or stopped if they made no difference to the health threat [16, 17]. This is important in relation to treatment adherence in paediatric rheumatology. Adherence to injectable treatments may represent a selection of coping behaviours, to manage the stimulus of symptoms, such as pain and loss of function. However, patients and their carers may appraise injectable treatment with experiences of distress, treatment anxiety, treatment failure and symptom return, leading patients and carers to change or adapt their coping behaviours, such as stopping treatment. Understanding experiences of using treatment, from social and cognitive perspectives, may provide opportunities to identify factors which influence behavioural adaptations leading to treatment withdrawal. These factors could then be used to target interventions to promote treatment adherence, improving health outcomes.

Evidence exploring treatment adherence in paediatric rheumatology may be out of date and therefore not reflect the modern everyday lives of children and young people (CYP) (defined as from birth to 25 years old),[18] parents and carers (PC) [19, 20]. More recent work has focused on psychological characteristics of parents and carers (such as impulsiveness and aggression) as well as other characteristics (such as the number of children being cared for) as important factors relating to treatment adherence [21]. However, this evidence used quantitative methods and artificially grouped participants into ‘good adherence’ and ‘bad adherence’ which may not reflect the dynamic experiences of everyday life of patients and carers (for example, having good adherence one week and bad adherence the next) [21]. This is further complicated as paediatric treatment adherence is behaviourally, socially and cognitively more demanding than adult adherence, as treatment demands can vary with children’s developmental stages as the child grows as the diseases progresses [22]. Further evidence of the modern, everyday lived experiences of treatment adherence are needed.

Previous studies exploring experiences of paediatric rheumatology, such as JIA [23, 24] reported parents and carers felt their children’s childhoods were ‘stolen’ and children with JIA felt different to other children without JIA. However, no studies have explored experiences of how children, young people, parents and carers with different rheumatological conditions ‘make sense’ of treatments and treatment adherence in their everyday lives. This is important, as many paediatric rheumatology services operate across sub-specialties, serving patients, parents and carers with a range of different rheumatological conditions, not just JIA. Much of the evidence is either historical or based on patient populations with only one disease, such as JIA, so further work is needed which explores everyday experiences of adherence holistically, across disease states, to provide evidence to design treatment services.

Aims

The aim of this study was therefore to explore children's, young people’s, parents’ and carers’ experiences of treatment in paediatric rheumatology services.

Aim, design and setting

In-depth semi-structured qualitative interviews with children and young people accompanied by parents and carers were conducted in a paediatric rheumatology service at the Great North Children’s Hospital (GNCH).[25]. A topic guide, based on themes identified through reviewing published academic literature was used to guide the interviews conducted between March 2023 and June 2023. Participants provided informed consent (if over 8 years old) or assent to take part in the study (if under 8 years old). Participants did not receive any payment, compensation or any other inducement to take part. Ethical approval for the study was given the Faculty of Medical Sciences Research Ethics Committee (Reference number 33823/2023) and the project was registered on the Trust’s Clinical Effectiveness Register.

Research Process

A convenience sample was recruited by one author (SS) who identified candidates for participation during routine clinical work. Candidates were given information about the project aims and processes. If candidates consented/assented, they were introduced to the other authors (KGP, APR) at their next routine hospital appointment. Two authors (KGP, APR) provided additional information, provided an opportunity to ask questions, assessed capacity to take part in research and took consent (or assent, if applicable). Interviews were conducted by one author (KGP), supervised by another (APR) with both the child or young person, alongside their parent or carer. Interviews were conducted in a private cubicle whilst the child or young person waited for/received parenteral treatment on a day-case ward. Interviews were recorded, transcribed, quality checked, anonymised and then deleted to protect confidentiality. Transcriptions were quality checked by reading through the transcript whilst listening to the audio recording to identify errors, which were subsequently amended by agreement of two authors (KGP, APR). Transcripts were anonymised by removing or replacing identifiable information such as names, places and other identifying characteristics. Participants were not contacted to verify the transcripts. Participants were recruited until theoretical data saturation (TDS) was reached (this is the point at which no further information was identified during interviews)[26]. TDS was identified through consensus by all authors.

Analysis

Analysis was completed by one author (KGP) under the supervision of the other authors (SS, APR). Thematic analysis was guided by phenomenological inquiry, whereby the noema (what is happening) and noesis (how it happens) are identified to describe the essence of a phenomenon [26, 27]. Data was primarily coded, and using a constant comparison approach, clustered and then grouped into themes [26]. Coding, clustering and thematic grouping were discussed at regular supervision meetings where analysis was interrogated to identify similarities and differences between codes to promote methodological rigour [26, 28]. Supervision meetings included at least three authors, drawing on expertise of qualitative social science, health research and clinical expertise. NVivo Version 12.4 was used to maintain an audit trail during analysis which was also reviewed during supervision meetings to improve credibility.

Participant characteristics

Data saturation was reached at 32 participants, consisting of 15 CYP and 17 PCs (see Table 1). CYP were between ages of 6 to 16 years, with an average age of 12 years. The average duration of the interview was 41 minutes and 43 seconds with a range from 22 minutes 4 seconds to 1 hour, 17 minutes. Juvenile Idiopathic Arthritis (JIA) was the most frequent diagnosis, with uveitis and joint damage being the most common comorbidity. The most common treatments the participants were using were infliximab, followed by tocilizumab and abatacept (see Table 2). Duration of treatment varied between a year and 10 years, with an average of 5.5 years.

Table 1

Summary of Demographic Data
Characteristics	Total n (%)
Children	15 (46.9)
Parents	17 (53.1)
Sex
Male	11 (34.4)
Female	21 (65.6)
Age
Children's Age, mean (range)	12 years old (6–16)
Diagnosis
Juvenile Idiopathic Arthritis	12 (80)
Juvenile Dermatomyositis	1 (6.7)
Chronic Recurrent Multifocal Osteomyelitis	1 (6.7)
Linear Scleroderma	1 (6.7)
Comorbidities18
Biomechanical musculoskeletal pain	3 (6.7)
Cataract	1 (6.7)
Chronic Fatigue	1 (6.7)
Glaucoma	1 (6.7)
Hypermobility	1 (6.7)
Joint damage associated with JIA	4 (26.7)
Lower limb length discrepancy	1 (6.7)
Obesity	1 (6.7)
Osteochondral defect associated with JIA	1 (6.7)
Uveitis	4 (26.7)

Table 2

Participants experiences of treatments in paediatric rheumatology
Current Treatment Plans n (%)
Infusions
Infliximab	5 (33.3)
Pamidronate	1 (6.7)
Tocilizumab	4 (26.)
Abatacept	4 (26.7)
Systemic Steroids	5 (33.3)
Rituximab	1 (6.7)
Tablets
Mycophenolate Mofetil	4 (26.7)
Azathioprine	1 (6.7)
Hydroxychloroquine	1 (6.7)
Injections
Methotrexate	2 (13.3)
Eyedrops
Dorzolamide/Timolol	1 (6.7)
Past Treatments
Infusions
Abatacept		1 (6.7)
Baricitinib		2 (13.3)
Infliximab		2 (13.3)
IVIG		2 (13.3)
Tocilizumab		3 (20)
Injections
Adalimumab		9 (60)
Etanercept		1 (6.7)
Methotrexate		11 (73.3)
Intra-articular Steroid Injections		11 (73.3)
Tablets
Mycophenolate Mofetil		1 (6.7)
Sulfasalazine		3 (20)
Eyedrops
Steroid Eye Drops		3 (20)

Themes

Findings demonstrated experiences of treatment in paediatric rheumatology appeared to oscillate between positive and negative trajectories mediated by somatic, social and cognitive factors. Somatic factors related to biological, physical, functional experiences such as feeling pain, joint swelling, or limited mobility. Social factors related to practical aspects of everyday life, such as missing school or work and relationships with people such as healthcare professionals or family and friends. Finally, cognitive factors represented psychological beliefs or responses described by participants. These factors appeared in participants’ experiences of diagnosis, treatment initiation and adherence. Thematic findings are grouped below into Somatic, Social and Cognitive factors. Each theme is described in detail below and exemplar data extracts to add further detail which are representative of the data are shown in Table 3.

Table 3

Data Extracts
Theme	Diagnostic Experiences	Treatment Experiences
Oscillation	“[We were] very confused [during the diagnostic process]. Because we were, so going down this avenue of rheumatic fever and everything [the diagnosis] was just changing all the time […], So I was feeling very overwhelmed and confused by it sometimes.” – P23, Mum of child with Juvenile Idiopathic Arthritis “It was scary [at the beginning], we’re that used to it that there's not a fear of it anymore, really, because we know what we're dealing with, but, obviously, my thought is how it's going to impact her in later life. How it's going to affect her going forward for jobs, and you know that, how she’s going to cope with certain situations, yeah.” – P29, Mum of Child with Juvenile Idiopathic Arthritis “And even now that I know, like what it is and what's happening, it's still very scary to see, like, what's going to happen to me like in the future?” – P12, Child with Juvenile Idiopathic Arthritis “Yeah, I think you kind of got a bit used to it. And you know now that if she has an illness [flaring], you probably know you’ve got to [go to the] hospital and she'll be in flares. She just -- It is still scary, but not how it was before. You get used to it; you do. Yeah.” – P20, Mum of child with Juvenile Idiopathic Arthritis	“It starts, looks good for a while. Stops, looks too bad. Start a new one, yeah, it goes all around the circle, starts again and again and again.” – P1, Child with Juvenile Idiopathic Arthritis “It's just never ending, so it's, you'll get used to a drug, and then we'll get put into a different one. It'll work for a little while and then it stops working, which just doesn't ever seem to keep anything under control? It's just very up and down all the time. Highs, lows, and it's just -- it just feels like it's never ending.” – P20, Mum of child with Juvenile Idiopathic Arthritis “Do I think she'll probably change medications again? Yes, I don't think she'll stay on this for very long. I think she'll end up coming off it again soon and going on to something else. [..] So, I think it's just– nothing definite, and that's how I kind of feel.” – P20, Mum of child with Juvenile Idiopathic Arthritis “She didn't tolerate it well at all. She was sick all the time and stuff. As soon as we come into the rheumatology team and told them, and asked what was happening [to her], they changed her drug.” – P31, Mum of child with Juvenile Idiopathic Arthritis
Somatic Factors	“Yeah, I felt like very scared because I went from like being able to, like, run around and play and then not being able to do anything.” – P12, Child with Juvenile Idiopathic Arthritis “Yeah, Well, it's just a condition, really, isn't it? It's -- I don’t know how to explain it. Felt a bit bad, having arthritis. But at the same time, it's a condition that could be controlled, from what I've read about it and now knowing bits and pieces about it, so it's not life threatening.” – P27, Dad of child with Juvenile Idiopathic Arthritis “Yeah, he does miss some school from joint pain. He restricted, like playing out, for the length of time he can play football, or the length of time we can go out for a walk, you know, he has to take multiple rests. I think he knows he's not the same as every other 14 year old lad, but there is worse out there. Do you know what I mean? So he still gets up, gives it a go, and then the next day, that's when it hits him [the pain]. So, the next day he'll be sore, or he'll be tired from whatever we did the day before.” – P18, Mum of child with Juvenile Idiopathic Arthritis	“I think it has been effective, it does work for times and she'll be fine. And at the moment, touch wood, her legs are fine because sometimes she's unable to walk, she's unable to get out of bed, and she can't even walk up and down the stairs. The medication does help and it does keep it controlled to a certain level.”– P20, Mum of child with Juvenile Idiopathic Arthritis. “It makes her well, just like any other normal [kids]. Yeah, she'll run round. She'll play.” – P25, Mum of child with Juvenile Idiopathic Arthritis He doesn't feel like it, really, it [the treatment] doesn't do anything. I suppose it does [work], cause they're saying that sometimes there's no active arthritis, so you know [it should work]. [...] I suppose it probably does, but from Johnny's perspective, he's probably not [feeling that it’s effective]. He doesn't really feel like much difference really.” – P18, Mum of child with Juvenile Idiopathic Arthritis “She's never really had any [side] effects from it. it's always been quite straightforward, which has been good, really.” – P16, Mum of child with Juvenile Idiopathic Arthritis “The side effects after the infusions were awful. I've never seen him that poorly like that in his life. That was really worrying though.” – P22, Mum of child with Juvenile Dermatomyositis “That made her upset, she was in a lot of pain with it, which obviously then made her frightened to have it. She was starting to hyperventilate because it was starting to hurt her. So, when that happened, it was like, ‘this isn't working’ now, [because] she's starting to really panic every time she's having that [medication], obviously we need another solution.” – P29, Mum of child with Juvenile Idiopathic Arthritis “It’s horrendous. […] She gets quite a lot of illnesses because of the immune suppressants. She had impetigo about 3 weeks ago and that's spread all over her face, so she needed antibiotics for that. So it does have a massive effect on her school, and she gets tired a lot as well.” – P20, Mum of child with Juvenile Idiopathic Arthritis
Social Factors	“[He’s] still doing his day-to-day thing, jumping on the trampoline, going out with his friends.” – P27, Dad of child with Juvenile Idiopathic Arthritis “Yeah, just followed what the doctor said. I mean, we didn't know too much about the condition to start off with.” – P28, Mum of child with Linear Scleroderma	Pro-treatment “Because the nurses told me that I'd be damaging him in a long term if [he] didn't have it, that was what they said.” – P18, Mum of child with Juvenile Idiopathic Arthritis. “He’s a big believer of what's the point in coming. But I'm -- I don't know. I'm kind of one of those people where I do what I'm told to do. And if the doctors told me to do something, then we have to listen -- and we have to do it because they're doctors.” – P18, Mum of child with Juvenile Idiopathic Arthritis “No, don’t think I am [worried about the side effects]. It doesn't affect that much day-to-day life, and she still managed to do everything that she wants to do. It’s not really an issue.” -- P28, Mum of child with Linear Scleroderma “They always let me change the hours [of my shift] when I need to get her, like the other week she was downstairs [the rheumatology clinic] [..] They stopped your treatment, and then we had to come back on that [medication] a couple of days later, but I was supposed to be at work that day, so we had to ring [my] work and say, ‘I'm not coming in, can I swap it [my shift], I'm going to come in another day,’ and they just say ‘that’s fine.’ ” – P28, Mum of child with Linear Scleroderma “We just take the letters in from the hospital and try to explain why [she was absent], I mean, we picked that school because they already had a child in there that has JIA.” –P31, Mum of child with Juvenile Idiopathic Arthritis. “I think, yeah, you definitely are looked after here. It's a really good hospital to be at, and you just know that they know what they're talking about.” – P16, Mum of child with Juvenile Idiopathic Arthritis “But we do have good family support, so if it's not me, it's his mum or your grandma or your sister or, you know, somebody in the family. So, we manage.” – P21, Dad of child with Juvenile Idiopathic Arthritis “Our work has been really supportive. They give us the time off to come in and bring Isla [to the hospital], so that's not an issue.” – P25, Mum of child with Juvenile Idiopathic Arthritis Anti-treatment “School, we've had big issues with school just because of his absence, so he's home schooled now actually. They were OK initially, weren't they, the school? But then I think as you got older, they just didn't [understand], Because you obviously had quite a lot of absences […]initially, they were fine for him to come in late if he was struggling, but then it just became more difficult and difficult [for school to be fine with it], and so as he grows older, I think they just expected him to just be ready to go every day and attend school. So now he's now being tutored at home.” – P21, Dad of child with Juvenile Idiopathic Arthritis. “Money's been a bit of a worry, especially mainly on travel. That’s the only thing really, I wouldn’t not come up here. I know she needs it, but it’s the money that [creates] worry the most.” – P31, Mum of child with Juvenile Idiopathic Arthritis “So, it’s a big chunk out of that sort of thing, that was one of the reasons why I don't work. We didn't know how often she would have to come to the hospital, whether she's having to go for physio, or where she's having to go for hydrotherapy. So, [it] takes time out of my day, whereas if I was working, I wouldn't be able to do that, because it would impact on my work life.” – P29, Mum of child with Juvenile Idiopathic Arthritis “Even at school, and like I say, some of the nurses didn't believe me, so I had to take photographic evidence of my own child being sick, which is not very nice. It literally took about two years to fight to get him off that medication,.” – P18, Mum of child with Juvenile Idiopathic Arthritis “It was quite difficult because, like I say, I didn't drive. His dad was always working, and his siblings were babies. So, it was quite hard.” – P18, Mum of child with Juvenile Idiopathic Arthritis
Cognitive Factors	“It's scary. When you're looking into it and what it is, it's just wondering how it is going to affect [her physically in] the long term...” – P31, Mum of Child with Juvenile Idiopathic Arthritis “[The diagnostic process] was long because we didn't know what was happening. And I think, if we'd had an idea of what it was, then we could have understood the process. But initially, we didn't know anything about it, and she was gradually getting worse, so we were totally in the dark right at the beginning.” – P29, Mum of child with Juvenile Idiopathic Arthritis “There's a certain amount of relief, I think, when you find out what it [the disease] is.” – P24, Dad of child with Juvenile Idiopathic Arthritis	“[We went to the psychologist] because of her methotrexate. The injections were really bad, and she was really struggling with the blood tests, and they suggested putting her into the psychologist here and we started to see her. We saw Charissa [Psychologist’s Pseudonym] for a little while, didn't we? And she just, talked to her, kind of try to get to the root of the problem and try to understand what it was. And she has got a lot better since we saw her.” – P20, Mum of child with Juvenile Idiopathic Arthritis “I think the methotrexate was the worst one. I hated that every week. It was terrible. I don't know [why], it was just a mental block because, like, I just couldn't press the button [for the self-injections] […] It's not that it hurts. It was just -- the feeling I just like, just knowing what was happening.” – P7, Child with Juvenile Dermatomyositis “Like the second time, I ended up screaming and crying cause I hated it so much because I developed a fear of needles.” – P2, Child with Chronic Recurrent Multifocal Osteomyelitis I think I just didn't like it because of the needles, really.” – P14, Child with Juvenile Idiopathic Arthritis “When we went to psychologist– and to be fair, they were fairly honest straight away, they said ‘we've seen the injections, we really don't know how we're going to get her to like them, but we’ll do what we can to like, try and give her choices.’ […] and she [Isla] actually just told me that she didn't feel like herself anymore and she didn't want to be here anymore. So that's when rheumatology [team] got involved and said, ‘Right. OK, then let's change everything [treatment regimen].” –P25, Mum of child with Juvenile Idiopathic Arthritis

Somatic Factors

Somatic factors described participants’ physical or biological experiences in paediatric rheumatology and were linked to both diagnosis and treatment. For example, participants reported physical manifestations of rheumatological disease led to somatic symptoms of pain and reduced mobility. These symptoms were also used as proxy measures to appraise the efficacy of treatment, where if a treatment improved symptoms it was considered successful, a positive trajectory. However, the return of symptoms or experience of physical side effects (such as vomiting) were perceived as treatment failure, a negative trajectory. If appraisal showed a negative trajectory, participants reports experiences of changing treatment, representing a turning point back to an upward or positive trajectory, whereby somatic experiences would improve (i.e. symptoms or side effects would improve).

Social Factors

Social factors were identified as experiences linked to social agents, such as family, friends, social institutions (like schools or hospitals) and social norms (such as ‘going to work’ or ‘out with friends’). Although social factors did appear linked to diagnosis as they appeared to represent ‘wellness’), social factors appeared more focused as a mode of appraising treatment adherence. These factors also seemed to oscillate between ‘pro-treatment’ factors and ‘anti-treatment’ factors. For example, participants reported being given information by healthcare professionals (such as doctors and nurses) in a child-friendly format, being made to feel cared for in the hospital, being given time off work or and having good family support networks supported treatment adherence. Participants also reported social factors which were linked with negative treatment appraisal, such as the cost of transport to the hospital, headteachers sending letters about school non-attendance linked to hospital appointments, and other family commitments.

Cognitive Factors

Cognitive factors were identified as participants’ reported feelings, emotions or strongly held beliefs which influenced experiences of diagnosis and treatment. Cognitive experiences linked to diagnosis were linked to fear of the unknown, which changed to relief when a decision was made about diagnosis. Fear was also identified in relation to treatment formulations (injectable) or specific medications (methotrexate). These cognitive factors were largely negative, though participants did report input from psychologists and counselling about treatment formulation and specific medications was positive.

What is pervasive in the data is an oscillation of experiences and trajectory whereby participants have experiences of negative somatic, social and cognitive factors until a ‘turning point’ at diagnosis and treatment initiation which led to positive experiences of somatic, social and cognitive factors. However, experiences appeared to ‘turn again’ to a negative trajectory during treatment, leading to treatment non-adherence, switching treatments, or further intervention with non-pharmacological support (such as psychological counselling). This oscillation between positive and negative trajectories of experiences appears to reflect the flare-remission-flare nature of rheumatological diseases more broadly.

Implications in context of existing research

Previous work has identified the importance of somatic experiences and social factors in paediatric rheumatology [29]. For example, physical somatic symptoms of reduced mobility and social disruption such as missing school, where considered ‘tipping points’ or health threats which stimulated visits to healthcare professionals for diagnoses. This echoes the findings above, where experiences of somatic symptoms and social factors led to treatment initiation and adherence. This also aligns to theory in the CSM, whereby somatic symptoms as well as social and cognitive factors, represent triggers, stimulus or ‘health threats’ which prompt behaviours such as visiting a health professional or starting treatment [17]. The findings presented above go beyond existing work by demonstrating somatic, social and cognitive experiences may be used to appraise treatment adherence behaviours by patients and carers in paediatric rheumatology.

This study adds to the literature as it considers the experiences of the paediatric rheumatology population holistically, rather than as individual conditions. Collectively this patient group represent a significant population [2, 3, 30] who access similar treatments, diagnostic pathways and health services. Understanding the relationship between somatic and social experiences and treatment adherence behaviours across this population enables targets for interventions to improve services to be identified at scale. For example, this study identified being able to ‘go to work’ or ‘out with friends’ were important social experiences which enabled patients and carers to appraise treatments positively. Existing work has identified the importance of understanding patient and carers values when making decisions about treatment [7]. The findings presented by this study, indicate the social value of treatment may be a factor influencing treatment adherence. Previous studies have reported the social values, though key parts of shared-decision making ideologies in healthcare, are reported to be missing from patient and carers experiences [31]. This means policy makers and practitioners must consider how social and cognitive values and preferences, such as how to access treatment (distance to the hospital, timing of the appointment, duration of the appointment) influences social lives of patients and carers when designing and delivering services.

Interventions which target social and cognitive factors which influence adherence are reported in relation to other disease states [32, 33]. These interventions adopt a social constructivist theoretical approach, to consider the intersection of systems of healthcare, family, finance, media and law which patients use to interpret and appraise their symptoms and treatment. In paediatric rheumatology, these could include integrating treatment times with social events for young people or carers or providing resources (like internet access, private spaces, work stations) for patients or carers to continue to work whilst waiting for their appointment. Alternative spaces to deliver treatments should also be considered, which could reduce the social disruption to patients and carers receiving treatments and increasing treatment adherence behaviours. Though of course this must be tempered with a clear understanding of the complication of treating paediatric rheumatology patients [34]. Further work is needed to explore broad social action which may also be required to educate employers, headteachers and others, to react flexibly to absence requests and help with costs of treatment socially and economically [8], reducing the social burden patients and carers face when trying to adherence to treatment.

Limitations of the study

A limitation of the study is interviews were conducted jointly with patients and carers, which may have influenced what participants were willing to say in front of one another. Additionally, recruitment of participants via the clinical team may have meant participants felt biased towards the clinical team (i.e., recruiting favourable participants). This sampling bias does not appear to have influenced the findings as these were not directly about the service being received, but rather broader experiences of treatment adherence. The work adopted standardised methods by using a topic guide, quality checking transcriptions, and reviewing the analytical audit trail which increases trustworthiness [28]. The methods used are reported transparently above and in sufficient detail for the study to be reproduced, increasing the dependability and credibility of the findings [28].The data was collected from a single site in North East England and the qualitative nature of the work means the findings are not generalisable however may be transferable to similar settings and conditions. Although the study identified factors which may influence treatment adherence in paediatric rheumatology, the work is limited as the depth or intensity of these experiences were not considered. Although this is likely to vary from patient to patient and carer to carer, further work is needed to explore the relationship between the intensity, depth and duration of experiences of somatic, social and cognitive factors influences treatment adherence.

The aim of the study was to explore treatment experiences of children, young people (CYP) and their parents or carers (PC) in a paediatric rheumatology service. The findings demonstrated patient and carer experiences of treatment adherence oscillated between positive and negative trajectories, influenced by somatic, social and cognitive factors. Further work is needed to explore the depth and intensity of these factors on treatment adherence and to identify interventions using these factors to support treatment adherence and improve health outcomes.

JIA Juvenile Idiopathic Arthritis

jSLE Junenile-onset Systemic Lupus Erythematosus

JD Juvenile dermatomyositis

UK United Kingdom

CYP Children and Young People

PC Parents and carers

Availability of data

The dataset supporting the conclusions of this article is included within the article and its additional files.

Acknowledgements

The authors would like to thank the participants for taking part as well as peer-reviewers who supported the publications of this manuscript.

Funding

No funding was obtained to conduct this study.

Author information

KGP was an MRes student working under the supervision of SS and APR. SS is a Paediatric Rheumatology Consultant in at the Great North Children’s Hospital, CLR is a Senior Lecturer in Pharmacy Practice at Newcastle University, and APR is a Lecturer in Clinical and Social Pharmacy at Newcastle University and Advanced Clinical Pharmacist at the Children’s Hospital.

Author contributions

SS, KGP and APR authors were involved in the design, development, delivery of this study. SS, KGP, and APR were involved in participant recruitment, data collection and analysis. All authors contributed to interpretation, drafting, writing and editing the manuscript. All authors read and approved the final manuscript and had access to the data.

Ethical declarations

Ethical approval for the study was given the Faculty of Medical Sciences Research Ethics Committee (Reference number 33823/2023) and the project was registered on the Trust’s Clinical Effectiveness Register.

Consent for publication

We confirm that all authors have approved the manuscript for submission. Also, I, Adam Pattison Rathbone, confirm that the manuscript has not been published, and have not been submitted for publication elsewhere.

Competing interests

The authors declare they have no competing interests.

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Editorial decision: Major revision
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Reviewers agreed at journal
21 May, 2024
Reviewers invited by journal
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A qualitative study exploring experiences of treatment adherence in Paediatric Rheumatology - Parents’, Carers’, Children’s, and Young People’s Perspectives

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