Article Text

Extended report
The patient perspective on remission in rheumatoid arthritis: ‘You've got limits, but you're back to being you again’
  1. Lilian H D van Tuyl1,
  2. Sarah Hewlett2,
  3. Martina Sadlonova3,
  4. Bev Davis4,
  5. Caroline Flurey2,
  6. Wijnanda Hoogland1,
  7. John Kirwan5,
  8. Tessa Sanderson2,
  9. Dirkjan van Schaardenburg6,
  10. Marieke Scholte-Voshaar1,
  11. Josef Smolen3,
  12. Tanja Stamm3,
  13. Maarten Boers7
  1. 1Department of Rheumatology, VU University Medical Center, Amsterdam, The Netherlands
  2. 2Nursing and Midwifery, University of the West of England, Bristol, UK
  3. 3Division of Rheumatology, Department of Medicine, Medical University of Vienna, Vienna, Austria
  4. 4Bristol Royal Infirmary, Bristol, UK
  5. 5Academic Rheumatology Unit, University of Bristol, Bristol, UK
  6. 6Jan van Breemen Research Institute | Reade, Amsterdam, The Netherlands
  7. 7Department of Epidemiology and Biostatistics, VU University Medical Center, Amsterdam, The Netherlands
  1. Correspondence to Dr Lilian HD van Tuyl, Department of Rheumatology, VU University Medical Center, 3A56, Amsterdam 1007 MB, The Netherlands; L.vantuyl{at}vumc.nl

Abstract

Objectives The aim of rheumatoid arthritis (RA) treatment is remission. As treatment should be targeted at outcomes relevant to patients, it is important to understand how patients perceive remission, and to assess whether the current definition of remission adequately reflects these perceptions. The objective of this study is to explore the patient perspective on remission in RA.

Methods Nine focus-group discussions in Austria, The Netherlands and UK were conducted, including patients in American College of Rheumatology (ACR)/ European League of Rheumatology (EULAR) remission, self-declared remission and in moderate/high disease activity. Moderators employed a prespecified interview guide helped to engage patients in a discussion on their experience with remission. Inductive thematic analysis was performed within each country, and identified themes were discussed across countries.

Results 47 RA patients (66% women, disease duration 9 years) participated. Three major themes of patient-perceived remission emerged: (1) symptoms would either be absent or strongly reduced, (2) impact of the disease on daily life would diminish by increased independence, ability to do valued activities, improved mood and ability to cope; (3) leading to a return to normality, including work, family role and perception of others. Patients felt the concept of remission was influenced by ageing, side effects of medication, comorbidities, accrued damage to joints and disease duration. Opinions on duration of state, the role of medication and measurement instruments varied widely.

Conclusions Patients characterise remission by the absence or reduction of symptoms, but more directly by decreased daily impact of their condition and the feeling of a return to normality. The next step is to study whether an additional patient-perceived measure of remission may add value to the ACR/EULAR definition of remission.

  • Rheumatoid Arthritis
  • Qualitative Research
  • Outcomes Research
  • Patient Perspective
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The degree of disease activity and response to treatment in rheumatoid arthritis (RA) are traditionally evaluated by outcome measures gathered in the RA core set or indices derived thereof.1–5 The core set contains three outcomes directly reported by patients, so-called patient-reported outcomes (PROs): physical function, pain and global assessment. These subjective patient-reported outcomes are at least as informative as other physical and biochemical (more ‘objective’) measures in assessing baseline disease status, improvement during interventions, or prediction of long-term outcome.6–10 However, several areas have only recently been identified as important to patients and, thus, as potential core areas for measurement. For example, data have shown that measurement of fatigue, one of the most important problems identified by RA patients, is highly reliable, sensitive to change and an independent determinant of disease activity.11–13 Therefore, OMERACT (an international scientific organisation studying outcome measures in rheumatology) now recommends fatigue should be assessed in all RA clinical trials.14 This reinforced the notion that patients should be regarded as crucial partners in obtaining relevant information and prioritising areas of research, as patients and professionals bring different skills, values and experiences to research.15 ,16

Treatment of RA is increasingly aimed at remission. In 2011, the American College of Rheumatology (ACR)/ European League of Rheumatology (EULAR) remission criteria were developed. According to these criteria, remission is reached when the tender joint count, swollen joint count, patient global assessment of disease activity and C-reactive protein are all smaller than or equal to 1, or when the simplified disease activity index is smaller than or equal to 3.3.6 The committee used prognostic factors and outcome measures available in clinical trial data, including the three core set PROs. PROs on other potential important aspects of remission were not available, highlighting the lack of knowledge on the patients’ perspective on remission. At OMERACT-10 patients and professionals agreed that there is an urgent need to study the concept of remission, including the identification and measurement of domains that are important to patients.17 This is in line with international recommendations for treating to target, with the target being remission and the treatment described as a shared decision between the patient and the rheumatologist.18

The aim of this study is to capture the patient experience of remission in RA.

Methods

Focus group discussions were conducted to investigate the domains of remission according to patients with RA in three European countries. Focus groups, rather than individual interviews, were used, as they promote discussion and debate among participants.19

Patients

Patients over the age of 18 years with a confirmed diagnosis of RA receiving usual care in one of the three centres (Reade Research Institute in Amsterdam, The Netherlands, Medical University of Vienna, Austria, and the Bristol Royal Infirmary in Bristol, UK) were asked to cooperate in a qualitative study.20 To get a broad range of patient characteristics, eligible consecutive patients attending outpatient clinics were invited to join a focus group discussion on ‘when your disease activity is as good as gone’ (rather than ‘remission’, which is not used with patients in The Netherlands and Austria, while UK patients are familiar with the word; this phrase was formulated with the help of patient research partners, who specifically advised us to refer to ‘disease activity as good as gone’, rather than ‘disease as good as gone’, as the disease cannot be removed, but the activity can.).

Initially, three focus group discussions were organised in every country: one with patients in ACR/EULAR remission, one with patients in self-declared remission and one with patients in a moderate to high disease activity state, defined by a DAS28 >3.2. Two-thirds of the patients in moderate to high disease activity must previously have experienced self-perceived remission or very low disease activity. Patients not fluent in the local language were excluded. Data collection was continued until saturation was reached. Three patient research partners were involved (WH, BD, MS-V).

Data collection

In the 2-week period before the focus group meeting, remission was assessed in all patients by the clinicians according to the new ACR/EULAR Boolean remission definition.6 Clinical data collection was limited to a 28 swollen and tender joint count, the laboratory measurements erythrocyte sedimentation rate (ESR) and c-reactive protein (CRP), and a physician global assessment of disease activity. At this same visit age, disease duration, experience with remission in the past, current self-perceived remission status, and the patient's global assessment of disease activity were recorded. Specifically, the following three questions were formulated: ‘Have you ever experienced a period of very low disease activity, in which you had the idea that your disease activity was as good as gone?’(yes/no) ‘Would you say that, at this moment, your disease activity is as good as gone?’ (yes/no), ‘Considering all of the ways your arthritis has affected you, how do you feel your arthritis is today?’ (10 cm visual analogue scale, anchors very well to very poor).

A common focus group interview guide was developed (see online supplemental file 1) and translated in the local language by a researcher from Vienna and The Netherlands. All focus group discussions were conducted by experienced qualitative researchers (MS in Vienna, SH, CF, TSa in Bristol and LvT in Amsterdam) who were not involved in the clinical care management of any of the patients. Discussions, lasting approximately 1 h, were taped and anonymised at transcription, with patient names being replaced by codes.

Analysis

Inductive thematic analysis was used to ensure that findings were grounded in patients’ data rather than imposed from professional literature. Initially, transcripts were read and reread to gain an understanding of, and familiarisation with, the issues. Then, small units of meaning were identified and given descriptive labels (codes). Next, the findings were explored to see how codes could be grouped to form subthemes, and finally subthemes were grouped to form overarching themes.21

This was carried out independently in each country by experienced qualitative researchers with their patient partners (TSt and MS in Vienna, SH, CF, TSa in Bristol and LvT and WH in Amsterdam). Next, the reports and supporting evidence from Vienna and Amsterdam were translated by two of the researchers in each centre, and all sets were shared. A teleconference between the qualitative analysers from each country was organised, followed by a 1-day meeting of all researchers and patients from all three countries, at which emerging codes and themes were compared and agreed.

All necessary documents were reviewed by an independent ethics committee under the responsibility of the chief investigator in each country. Patients were informed about the nature of the study, and they provided written informed consent.

Results

In each country, data saturation was reached after three focus group discussions, which resulted in a total of nine focus group discussions in total.

A total of 152 patients were approached to participate. As 105 declined the invitation, a total of 47 patients participated, including 66% women; overall mean disease duration was 9 years (table 1).

Table 1

Patient characteristics

Three main themes emerged (symptoms, impact and normality), underpinned by a theme of influential factors and a related theme of ‘assessment issues’. Together, they form the concept of remission according to the patients’ perspective (figure 1). The five themes are discussed below, including supporting quotes; subthemes and additional quotes are presented in tables 26.

Table 2

Quotes supporting the ‘symptom’ theme of patient-perceived remission

Figure 1

Main themes of rheumatoid arthritis remission. The three main themes (symptoms, impact and normality) are underpinned by a theme of influential factors, with an overarching theme of assessment issues.

Symptoms

It was important to patients that specific symptoms would either be completely absent or be reduced in intensity. Reduction of pain (Q1 and 2), stiffness (Q3 and 4), fatigue (Q5 and 6) and swelling (Q7) were mentioned frequently, but also recovery of strength (Q8) and improved sleep (Q9 and 10) were important to several participants (table 2).

Stability of disease was seen as a sign of remission, manifested as fewer flares, and decreased unpredictability and variation in signs and symptoms:UK2 G: For 5 full weeks, I've got out of bed and I've got on with the day, brilliant! I think that partly what I'm hoping is that being controlled means that there's a predictable component to my disease activity… that would be great because then if someone said to you they were wanting to have a party, you'd know you would be able to go. I've had to cancel so many social events.UK2 H: ‘I don't know how I'll be, If I'm okay I'll come.’UK2 G: Yeah, but if your disease was controlled you'd be like, ‘Yeah, I'll come along, I'll leave by ten and I'll probably drink too much before then!’ That would be good. That's a major event.

There was no agreement on the importance of severity of symptoms. For some patients, remission meant the complete absence of the symptom (Q1), while for others, a certain basic (low) level would suffice:NL2 3: well, the pain, purely the pain. That's the whole story. The other day someone said to me ‘girl, you don't know how it feels to live with pain everyday’. Then I said, ‘in fact I do’. When you have arthritis, you always have pain. Even in good periods. There is always something, when you get up you think, ‘oh yes’. But you accept it…MOD: even in good periods?NL2 1: yesNL2 3: You accept it, because you got the disease.

Impact

Reduction in the impact these symptoms led to was deemed as important as the symptom reduction itself. These impacts included physical functioning (Q11), activity of daily living (Q12 and 13), and being independent (Q14 and 15) (table 3). Patients saw remission as being able to do valued activities, have an improved mood and feel able to cope.UK3 L: I can just get on and do whatever I likeAT3 5: I realize, when I am absolutely without pain and there is no severe swelling, one feels somehow more positive, … well I realize that I am active, I make a lot of plans and I am somehow nicer to my colleagues and co-workers and just agreeable. And my husband is happy as well.

Table 3

Quotes supporting the ‘impact’ theme of patient-perceived remission

Normality

The reduction in symptoms and impact of the disease on daily life would eventually mean a return to normality.UK2 H: So, um, but for me remission is, like you say, feeling normal really.NL1 C: When I am doing well, I am my normal, cheerful self.

Return to normality could either be a return to the ‘old’ normal life, or a ‘new normality’ including the ability to (regain) work (Q16), enjoy one's family role (Q17 and 18) and be seen as normal by other people (Q19 and 20) (table 4).

Table 4

Quotes supporting the ‘normality’ theme of patient perceived remission

Assessment issues

There was no consensus on the minimal duration of this state that was needed in order to define it as remission, which varied from 24 h to forever (Q21–24). Likewise, the opinion on role of medication in the concept of remission varied between patients (Q25–27). There was confusion over the patient global measure, which was felt to be inappropriately worded to capture remission as it did not ask about disease activity (Q28); moreover, patients felt that the score was heavily influenced by their mood that day (Q29 and 30). Patients varied in the level of symptom/impact they thought necessary to achieve remission (eg 0–4 swollen joints), and the ACR/EULAR remission criteria (discussed in the UK groups) were perceived as too stringent (Q31). By contrast, others were disappointed with the symptoms they still experienced while in clinical remission (Q31 and 32) (table 5). Ideas on measurement of remission focused on measuring what one could do, rather than how one felt.AT2 2: When both of us try to open a bottle of mineral water—the same bottle—then we have the same starting point. We cannot open the bottle because the disease is active, we are not strong enough to open it. And actually this is the criterion, isn't it? This is better than a line with a mark [VAS], because that is only a subjective opinion.

Table 5

Quotes supporting the theme ‘assessment issues’ of patient perceived remission

There was a belief that the patient and physician definitions of remission might differ:NL1 F: So when you ask me if my disease is as good as gone... Well, my physician says it is. But if you ask me, do I feel better? Well no, I am not cured. My life, my days, my weekends are focused towards: ‘maybe I need to sleep’; or ‘if we do this on Saturday, I will need to rest on Sunday’; my grandmother of 80 years old can do more than me sometimes…

Influential factors

Patients struggled with the concept of remission itself, feeling it was confounded by normal ageing (Q33), side effects of medication (Q34 and 35), symptoms from comorbidities (Q36), accrued damage to joints (Q37) and disease duration (Q38) (table 6).NL3 1: It is sometimes difficult to say, the fatigue for example: age, medication or arthritis? But the symptoms are very specific, wrists and stuff. If you strain them, it really hurts. That is arthritis.

Table 6

Quotes supporting the ‘influential factors’ theme of patient perceived remission

Discussion

This is the first study that gives insight into the perception and experience of remission in patients with RA. Patients indicate that, for their disease to be as good as gone, the symptoms would need to be absent or reduced in intensity, but more in the foreground was the reduced impact of these symptoms on their life, eventually leading to a feeling of normality. This is consistent with the proposal that patients experience the impact of their condition, an interaction between severity of the disease, their personal circumstances and their ability to cope with the condition, and are less directly aware of specific pathological changes.22

Patients struggled with attribution of signs and symptoms to either the disease or influential factors, such as aging or side effects of medication. Many PROs assume that patients are able to differentiate between, for example, pain due to RA and pain due to flu, and there is some evidence that this may well be the case.23 Conceptual aspects of remission that are of great interest to professionals, were hard to grasp for patients; opinions on importance of duration for defining remission, as well as on the use of medication were very diverse; if anything, these aspects should be reviewed for each patient individually. In this respect, the omission of duration as well as medication from the ACR/EULAR remission definition seems sensible.

Not much is known about the patients’ perspective on remission. The only comparable work is a focus group study by Kristiansen et al, studying self-identity, social relationships and work relationships of early RA patients in remission.24 As participants were chosen because of the success of the treatment regimen they were taking, they had experienced only limited changes in everyday life, and were able to manage the varying limitations experienced. Interestingly, aging was identified as a means to normalise symptoms and disabilities, which was also seen in our study.

More research has been done on perceptions of patients in active disease, that shows a lot of resemblance with our work on remission. A recent cooperation between patients and professionals resulted in a new patient-derived score to capture impact of RA on daily life (RAID).25 The domains captured by the RAID, that is, pain, daily functioning, fatigue, sleep, physical and emotional well-being and coping, were all of importance to the perception of remission as well, although patient-perceived remission may not necessarily be represented by a low score on the RAID: RAID specifically measures impact of RA, while remission is a larger concept, where low impact leads to a return to normality, which is a complex concept in patients with a chronic disease.26

The new definition of remission in RA has been hampered by the lack of adequate data on the patient perspective of remission. The PRO included in the new remission criteria, the patient global assessment of disease activity, is a well-known and widely used instrument that should give an overall rating of the patients’ perception of disease activity. However, cut points for remission have never been validated and recent studies suggest that a remission PtGA of ≤1 might be too restrictive, excluding patients with comorbid conditions to achieve remission.27 ,28 Although not specifically addressed in this study, our work suggests that the PtGA is interpreted differently by different patients and can be influenced by many aspects of the patients’ life. As such, the PtGA may reflect disease impact, not just the notion of pathological severity implied by ‘disease activity’. Our study provides domains of patient-perceived remission that may contain information currently not captured by the ACR/EULAR remission criteria, yet crucial for optimal targeted therapy, thus implying that both should be measured.

This study had a qualitative design, ensuring a wide, unbiased approach towards the identification of aspects of remission according to patients. The involvement of patients from three different countries and different languages, allows for transferability across western Europe. Another important point is the involvement of three patient research partners who ensured a patient perspective during every phase of this work, through their input at the development stage of this project, by reviewing the patient information material and interview guide, by being an observer during the group discussions, as well as taking part in the interpretation and analysis of the data and, finally, by their participation in the meeting of researchers in Amsterdam.

A limitation of this study is that the Dutch and Austrian transcripts were analysed locally, that is, not translated and reviewed by the other centres. However, the data were carefully combined during a meeting with researchers and patient partners. For example, it was discussed that not all aspects of the domains were mentioned in every country (improved mental power was only mentioned in Austria; swelling was hardly mentioned in The Netherlands). During the meeting of researchers it was decided that these aspects would nevertheless be taken to the next phase of the study.

The intention to have three group discussions stratified for disease activity was not strictly effectuated. As patients were invited to join a specific group several days prior to the actual day of the discussion, disease status could have changed in the meantime; this was especially the case for the ACR/EULAR remission group, where the patient global assessment on the day of the discussion caused a shift in classification in two patients. This resulted in a total of 13 patients that were actually in ACR/EULAR remission. However, this was compensated by the number of patients who had experienced periods of remission and were able to share their experience. Moreover, it was never our intention to analyse the results of the three groups separately; the stratification was made to ensure a variety of conditions and experiences, yet grouping patients in a similar condition together to enhance group dynamics.

It can be debated whether the patients who participated in the group discussions are somehow different from the patients who declined the invitation. In each country, the group in ACR/EULAR remission was the most difficult to compile. This was most likely due to the low prevalence of ACR/EULAR remission, but also due to this group of patients feeling well and working/not wanting to think of their disease.

There were clear signals that factors, such as age, disease duration and comorbidity play an important role when evaluating remission perceptions of patients. However, this study was not designed to specifically address these issues, and the qualitative nature does not allow for stratification of results. Quantitative research would be needed to test this. Therefore, follow-up research has been initiated, where all domains will be rated for importance by a second larger group of patients. This work will also include identified influential aspects of age, drug side-effects, damage and disease duration. The most important domain(s) of patient-perceived remission, which will most likely relate to the overall impact of their condition, can then be taken forward in validation studies, to evaluate their psychometric properties, and added value to defining remission in RA.

In summary, the patients’ perspective on remission in RA is characterised by the absence or reduction of symptoms, by decreased daily impact of their condition and the feeling of a return to normality. The next step is to quantitatively study the identified domains and their added value to the ACR/EULAR definition of remission.

Acknowledgments

This study was supported by a research grant from the European League Against Rheumatism

References

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Footnotes

  • Handling editor Tore K Kvien

  • Contributors All authors significantly contributed to conception and design, acquisition of data/analysis and interpretation of data, of drafting the article or revising it critically for important intellectual content and approved of the final version published.

  • Competing interests None.

  • Ethics approval The medical ethical committee of the VU University Medical Center, of the University of Bristol, and of the Medical University of Vienna.

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

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