It s not just the joints, it s the whole thing: qualitative analysis of patients experience of flare in psoriatic arthritis

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1 RHEUMATOLOGY Rheumatology 2015;54: doi: /rheumatology/kev009 Advance Access publication 9 March 2015 Original article It s not just the joints, it s the whole thing: qualitative analysis of patients experience of flare in psoriatic arthritis Anna R. Moverley 1, Karen A. Vinall-Collier 2 and Philip S. Helliwell 1 Abstract Objective. Flare is a commonly used term in arthritis, including PsA, but remains undefined. This study seeks to explore patients experience of flare. Methods. Eighteen semi-structured interviews were undertaken with patients with PsA in an informal clinical setting. Patients were purposively sampled from specialist clinic lists to reflect the major elements of the psoriatic disease spectrum. Approximately one-third of patients reported themselves to be in a flare state at the time of the interview. Transcripts underwent thematic analysis according to the recommendations of Braun and Clarke (Using thematic analysis in psychology. Qual Res Psychol 2006;3:77 101). NVivo software was utilized, with initial coding reflecting the source data, such as pain, feeling miserable and feeling slowed down. Codes were then grouped into associated clusters. These clusters and the interrelationships were then summarized into emergent themes. Results. The mean age of the patients was 44 years, and patients average disease duration was 4.5 years. Nine main overarching themes pertaining to flare were identified: physical symptoms, social withdrawal, psychological symptoms, fatigue, loss of normal function, triggers, management of pre-flare, management of flare and timing. These show some degree of overlap and are interlinked, with some degree of temporality emerging as related to patients experience of flare. CLINICAL SCIENCE Conclusion. This study has identified a number of components of flare in PsA from the patient perspective. Although the emergent themes are of an overlapping and interactive nature, it is clear that flare in PsA is more than a swollen or tender joint count as measured in clinical practice. Key words: disease flare, psoriatic arthritis, qualitative research, patient perspective, symptoms. Rheumatology key messages. Flare in PsA is more than a tender and swollen joint count. Patients with PsA experience a wide range of symptoms while in the flare state. Further work needs to be done to identify key flare domains in PsA from the physician s perspective Introduction PsA is a chronic, disabling condition that affects up to 30% of people with psoriasis. The spectrum of disease is wide, involving joints, skin, nails, enthesitis, dactylitis 1 Leeds Institute of Rheumatic and Musculoskeletal Medicine (LIRMM) and 2 Academic Unit of Health Economics and Dental Public Health, University of Leeds, Leeds, UK Submitted 16 April 2014; revised version accepted 20 January 2015 Correspondence to: Philip S. Helliwell, Leeds Institute of Rheumatic and Musculoskeletal Medicine (LIRMM), University of Leeds, 2nd Floor, Chapel Allerton Hospital, Harehills Lane, Leeds LS7 4SA, UK. p.helliwell@leeds.ac.uk and spinal disease. Patients with PsA often describe a flare of their disease. However, unlike in other inflammatory arthritides [1 8], there has so far been little work on establishing a definition of flare in PsA, although a handful of existing articles provide varying descriptions, incorporating some to almost all aspects of the disease spectrum [9 12]. In contrast to the lack of current work in PsA, there is a growing wealth of information as regards flare in RA. The OMERACT group initially identified that patients did not consider synovitis an essential part of flare; in fact, many experienced flare as a much more widespread collection of systemic symptoms [3]. Recent international focus group work concludes that flare can be! The Author Published by Oxford University Press on behalf of the British Society for Rheumatology. All rights reserved. For Permissions, please journals.permissions@oup.com

2 Patients experience of flare in PsA experienced as a complex, multi-layered, whole-body experience [2]. Since it is the patient who experiences the flare of disease, we propose to establish a patient-driven definition of flare in PsA by eliciting the patient perspective. Qualitative research methods provide the ideal methodology for this type of study [13]. Further, a patient-defined definition is essential to fulfil the face validity section of the OMERACT filter [7]. Flare in PsA has been variably described to date as absence of remission [10] or absence of minimal disease activity [12], both of which refer to not meeting a set of predefined disease activity criteria. A further definition of psoriatic disease flare is provided by Kivitz, in which flare is defined as a part inversion of an ACR20 response [9]. However, it has been shown in RA that patients may require a small change in disease state to consider disease as worsening, but large changes to note disease as improving, as the intensity of change is not symmetrical between the two extremes [1]. Only one article in the literature refers to a self-reported flare in PsA, and this is given no further qualification [11]. There are very few qualitative studies in PsA [14]. Existing work tells us that concepts important to psoriatic patients are not well measured by available instruments [15], and even basic concepts of disease activity are not adequately covered by existing measures the PsA Quality of Life measure (PsAQoL) and the Psoriatic Arthritis Impact of Disease questionnaire (PSAID) being the only standardized PsA-specific tools [16, 17]. There is also a discrepancy between physicians and patients measurements of disease [18]. One paper alone has explored the patient experience of living with PsA [19], which is described as an ongoing, dynamic process of balancing [19]. Therefore, a need has been identified and more work is required in this area. In this article we seek to explore patients experience of flare, and management strategies involved in coping with disease flare as and when it occurs. Methods Patient selection and interviews This study was carried out at a tertiary care Rheumatology unit in England. Local ethics committee approval was obtained (12/YH/0041) and all patients gave informed written consent according to the Declaration of Helsinki. Patients were purposively selected from clinic lists by A.R.M. and P.S.H., with assistance from clinical nurse specialists in rheumatology, to include those who had experienced recent or previous significant peripheral disease flare and to reflect a range of disease characteristics (oligoor polyarthritis), age, gender, disease duration and medication. We also chose to interview a number of patients with significant axial disease (mean BASDAI 7.1) or skin burden [mean Psoriasis Area Severity Index (PASI) 4.6]. These patients again reflected a range of age, gender, disease duration and medication. Patients were approached by telephone or at a routine clinic visit by A.R.M., and those interested in participating chose appropriate appointment times to attend for interview at a later date. All patients fulfilled the classification criteria for PsA (CASPAR) for PsA [20]. A.R.M. conducted a total of 18 semi-structured face-to-face interviews with patients in an informal clinical setting, each lasting between 25 min and 2 h. In line with recommendations [13], there was no rigid procedure for interviewing, and a topic guide (see supplementary data, available at Rheumatology Online) was used only as a guideline. It was not exhaustive, and new topics generated by the participant were used to inform and update further interviews in keeping with a method of constant comparison. Analysis All interviews were audiorecorded and transcribed verbatim, and each transcript was verified by A.R.M. NVivo software was utilized (nvivo version 9.0!, QSR International, 2012). Thematic analysis may be used for identifying, analysing and reporting patterns (themes) within data and is described by Braun and Clarke [21]. In accordance with their guidelines, the transcripts were initially read, re-read, annotated and coded by A.R.M. in consultation with K.A.V-C. (phases one and two of analysis). Similar ideas brought up by different patients were studied in greater detail, leading to the identification of key candidate themes. Subthemes and overarching themes were subsequently developed and refined by discussion between A.R.M. and K.A.V.-C. to complete phase three. All candidate themes were then reviewed, and redundant themes were explored and discounted as appropriate, as outlined in phase four of analysis. Final themes were agreed and named and the report produced (phases five and six). Negative case analysis was also undertaken. Results Patients The mean age of the patients was 44 years, and the average disease duration was 4.5 years. Approximately onethird of patients (n = 5) reported themselves to be in a state of flare at the time of the interview (Table 1). The data collected in the interviews were subjected to thematic analysis as outlined above in order to gain a sense of the predominant or important themes. The themes that were identified, categorized, coded and analysed therefore serve as a reflection of the entire dataset, thus offering a rich overall description. Nine main overarching themes pertaining to flare were identified and showed interaction as well as some overlap, as illustrated in Fig. 1. Unsurprisingly, physical symptoms emerged as a significant burden in patients experience of flare. This theme included such nodes as pain, heat, stiffness, swelling and psoriasis. It s blooming well sore, you could fry an egg on it (patient 18) was initially coded as nodes heat and pain, two of the subthemes under the umbrella of physical

3 Anna R. Moverley et al. TABLE 1 Patient characteristics and flare state at time of interview Pseudonym Gender Age, years Disease duration, months Currently in flare Significant skin burden Significant axial burden Interview length, mins:s Patient 1 M N N N 29:35 Patient 2 F Y N Y 49:27 Patient 3 F Y N N 59:04 Patient 4 M N N N 43:14 Patient 5 M N N N 64:32 Patient 6 F N N Y 30:37 Patient 7 F N N N 23:57 Patient 8 F N Y N 36:41 Patient 9 M Y N N 35:32 Patient 10 F N N N 76:16 Patient 11 F Y N N 27:09 Patient 12 F Y Y N 44:36 Patient 13 M N N N 25:16 Patient 14 F N N N 55:20 Patient 15 M 30 6 N N Y 28:54 Patient 16 M 24 8 N Y N 23:46 Patient 17 M N Y N 24:31 Patient 18 M N N Y 38:59 N: no; Y: yes. FIG. 1Thematic diagram of overarching themes pertaining to flare symptoms. In some cases, the psoriasis would be a significant part of the flare. When the flare comes, when I get my flare up, I get my psoriasis pretty bad (patient 8). Symptoms were often multiple at any one time, so that patients experienced a number of aspects of physicality in combination, for example, swelling, throbbing, heat and stiffness. The finger wouldn t bend and I felt it was the swelling that was stopping it bending. It was swollen from the knuckle on the hand to that knuckle there, totally swollen. It throbbed all the time. It wasn t so much painful as a hot throbbing sensation, and as I said it was hot to the touch, very hot to the touch (patient 7). Fatigue also emerged as another significant burden in patients experience of flare. Despite having a lot of commonality with physical symptoms in the descriptors used by patients, it was such a dominant theme, seen across all participants, that it warranted differentiation from other physical symptoms; patients complained of feeling tired, battery gone and being flu-like....and [I] keep myself going but it s really difficult because the tiredness just takes you over, takes over...it is hard. It s hard because you can t keep your eyelids open, you re fighting it (patient 11). It was almost like I needed plugging in like a mobile phone, the battery had gone (patient 13). It s the fatigue...and it s just absolutely, it s like having flu but not having flu, it s absolute bone weariness (patient 18). Patients also experienced a variety of other related symptoms such as lack of motivation and loss of appetite, as linked to fatigue, while experiencing disease flare:...this kind of feeling of just being generally unwell and not being able to motivate myself... (patient 14). You ve seen me at my worst, and I lost a stone in weight and I ve never been that thin (patient 12). Patients noticed a significant loss of normal function while in the flare state: I couldn t even brush my hair (patient 10). In addition to loss of independence and increased dependence on others, patients noted loss of movement and loss of hobbies. I couldn t function. I couldn t function at all, and I m used to being active and doing lots of stuff, and then I couldn t do anything. I hardly left the house for months (patient 1). When it s bad l ll

4 Patients experience of flare in PsA say, Right you re going to have to do the shopping yourself...can you change the bed, I can t grip... (patient 8). Psychological symptoms included frustration, depression, embarrassment and fear. Having to depend on other people to do things for me I find really frustrating (patient 11). Fatigue and loss of normal function were cited by patients as contributing to some of the psychological symptoms they described, highlighting the impact of these themes upon the patient. I know I mentioned this before, but the mental health side, the tiredness that that causes and the frustration, and I think if you asked a question about how frustrated do you get with your arthritis most people would put a 10 (patient 2). I can t say I was bothered for [the wedding], which is a great shame because, looking back, I ve missed out on an awful lot (patient 13). A further knock-on effect of the physical symptoms, and indeed the psychological symptoms, was the social impact, which was significant for a number of patients experiencing disease flare. Patients noted loss of social life, not wanting physical contact and social withdrawal. [It has a knock-on effect]...on my work, my marriage, my poor husband. He s been fantastic, but it does affect me, what I want to do at home. I can t be bothered to do anything. I won t want to go out. I like to do sporting activities, but I can t do them. I know that I want to keep healthy, but I can t go and do what I want to do (patient 12). These first five overarching themes relate to the patients experience of the flare itself, i.e. an overwhelming collection of physical, psychological and emotional symptoms that clearly interact. There is a significant psychological element to the physical symptoms, in addition to the psychological aspect of the disease in its own right, and the fatigue element crosses these two subdivisions. There is, as a result, a significant loss of function and social interaction, which in turn has a huge impact on the patient s everyday life, much more than might be expected from a painful or a swollen joint or an area of skin disease. What was initially understood by the research team to be patients understanding of flare was further classified following the creation of a thematic diagram. The following four themes have an added dimension of temporality and can be best understood in terms of thinking about the directions/interactions outlined in Fig. 1: triggers, timing of flare, management of pre-flare and management of flare. Patients noticed particular triggers for flare, even if not recognizing a pre-flare state, including such as if it was damp weather it was definitely worse (patient 1). [If I] have been doing a lot of physical work, that would be the main question to ask, because, more often than not, that s what sets it off (patient 15). I know that if I ve done something, like...lifted a box the other day...i will know next day I will be ok, but the day after I will be hurting... (patient 2). The node timing of flare included duration of flare, which is variable:...you can tell the difference...i know it s not going to flare up to a bad attack, whereas...when I wake up if it s really severe I know...so you can tell the degrees of the flare up (patient 12). Sometimes it could just be a few hours...sometimes it could last all day, maybe a few days (patient 17). Management of pre-flare included the node warning of flare: I know through the night that it s going to be a bad day because I get up and down constantly (patient 2). This understanding of disease appears to come with greater disease experience, and can be seen more clearly in patients with longer disease duration. For example, patients with short disease duration would tend to cite their flare as having no warning. There s no kind of like...the day before you feel a bit more achy (patient 16). Those with many years experience would notice things more readily: I didn t know what was happening in the first one...[now] the minute it starts to be more than my hands and wrists, it starts to ring alarm bells (patient 14). Patients also developed strategies for management of early pre-flare once recognized: I ll just say to him, Right, I m having a lazy day today....i get the burning sensation, and then I think, Right, just calm down now, put them down and leave things alone. And I do know when it s coming, yes (patient 8). I just have to be right gentle with myself and understand that I have to do things, and I have to move, and that I just sort of, I m gentle (patient 18). Personal adaptations form part of the management of the condition, and patients tend to adapt activities to suit their current disease state. The management of flare theme expands on this idea and includes nodes such as self-medication and self-help, resting, avoiding things or, alternatively, just getting on with it. Patients also noted a flare could be managed by trying to understand it and by seeking medical attention. I try to understand the disease, and I try to help myself as much as I possibly can...whether it s doing the physio[therapy] exercises every day or reading up on things or trying to maintain a reasonable diet (patient 3). When the pain is that bad and I can t stand it anymore, and I ve taken painkillers and anti-inflammatories and nothing seems to be working, and then I think it may be time to go that bit further and come down and have the steroids...so then I m fine for a good six months or so (patient 11). Patients appear to recognize the flare state earlier once disease is established, and management also reflects this. Patients learn to self-manage, and avert the full flare in many cases. Recourse to medical help occurs when all else has failed. Discussion Patients describe a variety of symptoms in relation to a flare. Physical symptoms occur in combination, but there are also many other aspects of a flare, such as fatigue, psychological dysfunction, quality of life issues and loss of normal function. Flare in PsA is a lot more than a swollen joint count as measured in clinical practice. The management of flare theme gave insight into the patient journey through flare and the learning experience

5 Anna R. Moverley et al. that patients gain from disease duration. Patients early in the disease course identify a pre-flare state, and, with time, learn to manage triggers and a pre-flare in order to prevent progression to the full flare situation by recourse to self-management strategies and early medical intervention, as outlined above. It is only when this approach fails to halt the flare s progression, or when the flare becomes out of control that a full flare may emerge, requiring more definitive action. While data saturation was reached by patient 11, negative case analysis identified two noticeable exceptions regarding the patients journey through management of flare. This prompted further interviews and exploration to help differentiate whether these were indeed isolated cases or more common within the population. First, patient 3 unexpectedly described a permanent flare state, having reached the end of the treatment line, which was a new concept for us. In this situation, the patient journey has effectively failed, as there are no therapeutic options to curtail flare. Second, the patient s journey also fails if a patient has no warning of flare or understanding of triggers (patient 7), so that there is no option to pre-empt the inevitable occurring. Overall, however, it has become clear that there are grades and stages of flare that patients learn to manage, and patient education becomes ever more useful with advancing disease duration. This may be explored further in future work and have clinical implications in terms of patient education. In contrast to our work in PsA, current work in RA has concentrated more on the breakdown of flare into different stages [2, 22]. Flurey s work [22] outlines steps whereby a patient moves from daily life through pre-flare and flare to unmanageable flare. Hewlett et al. [2] describe six stages of flare: symptoms and early warnings, self-management of intensifying symptoms, defining as uncontrollable flare and seeking help when symptoms cannot be contained, contributed to and underpinned by the individual context and uncertainty. Although the shape of the thematic diagram is different from ours, the underlying base components of flare are similar in the two different diseases. In Hewlett s work [2], patients in RA flare describe joint pain, swelling, heat and stiffness as part of symptoms and early warnings. Although these are all important components of our physical symptoms theme, there is also the added significant dimension of skin disease in the work presented here. Patients in flare of both diseases describe a profound compromise in function, a loss of participation, social withdrawal, emotional distress, and fatigue. Some, but not all, patients in both conditions experience an early warning of an impending flare. In contrast, systemic features are listed as a major issue in rheumatoid flare [2], whereas in a psoriatic disease flare these features were subsumed into themes of physical symptoms and fatigue. Patients in rheumatoid flare describe reduced cognition and lack of concentration, but in our work these features were covered by the overarching themes of fatigue and loss of function. Disturbed sleep is listed separately as a feature in rheumatoid flare, but is subsumed into the fatigue theme in our work. In summary, excepting skin symptoms, components of disease flare are similar in the two conditions but their weight is different. In both conditions, patients will attempt to selfmanage intensifying symptoms, define an uncontrollable flare and seek help when symptoms cannot be contained. It is indeed true for PsA as well as for RA that there is a depth and breadth of symptom intensity and consequences that extend beyond a count of painful, swollen joints [2]. Strength and limitations The strength of our paper lies in the grounding of the research within the patients accounts by using a qualitative approach. A possible criticism is that the interviews were undertaken by a clinician, which may have created a more medical agenda for the discussion topics, although no ongoing doctor patient relationship existed at the time of interview. The interviews were also conducted within the hospital setting, although outside the clinic setting, which may have impacted upon patients full disclosure of the nature of their flare and preferred management processes. We had considered that a relationship between peripheral disease flare, skin disease, axial disease and other aspects of flare might have emerged, but although one patient noted that a joint flare would always be preceded by a skin flare, we did not have enough material to explore this further. Funding and ethical approval permitting, it would have been beneficial to adopt a more grounded approach to interviewing, allowing patients to be approached across different National Health Service trusts and outside the specialist clinic in our tertiary centre, thereby accessing a range of disease experiences, progression and care pathways. However, this study did sample a good patient range of disease characteristics, age, gender, disease duration and medication. Sampling from the tertiary setting alone could explain the high percentage of patients currently experiencing flare, but this has most likely had a positive influence on the findings in terms of reducing recall bias, as fewer patients were describing a previous and subsequently settled flare. Clinical and research implications This study has identified that a flare in PsA is much more than a swollen joint count or a worsening of psoriasis. A number of PsA-specific composite measures have been developed [23, 24], based on the core set of outcomes for PsA proposed at OMERACT-8 [25]. One limitation of these indices, possibly because of minimal patient involvement in their development, is that they may not address areas of disease activity important to patients, including fatigue, psychological impact and social withdrawal. We propose, therefore, to take the prominent nodes and emergent themes from our data set towards a formal definition of flare by undertaking a patient and physician Delphi to identify core domains of flare in PsA. The ultimate goal of such a process would be the development of a PsA flare tool for use in both clinic and clinical trials

6 Patients experience of flare in PsA Acknowledgements The authors would like to thank the participating patients for their involvement in this study. A.R.M. was funded in part by a grant from Arthritis Research UK. Funding: This study was part funded by The Psoriasis and Psoriatic Arthritis Alliance, UK. Disclosure statement: The authors have declared no conflicts of interest. Supplementary data Supplementary data are available at Rheumatology Online. References 1 Berthelot JM, De Bandt M, Morel J et al. A tool to identify recent or present rheumatoid arthritis flare from both patient and physician perspectives: the FLARE instrument. Ann Rheum Dis 2012;71: Hewlett S, Sanderson T, May J et al. I m hurting, I want to kill myself : rheumatoid arthritis flare is more than a high joint count an international patient perspective on flare where medical help is sought. Rheumatology 2012;51: Bingham CO III, Pohl C, Woodworth TG et al. Developing a standardized definition for disease flare in rheumatoid arthritis (OMERACT 9 Special Interest Group). J Rheumatol 2009;36: Bingham CO III, Alten R, Bartlett SJ et al. Identifying preliminary domains to detect and measure rheumatoid arthritis flares: report of the OMERACT 10 RA Flare Workshop. J Rheumatol 2011;38: Kett C, Flint J, Openshaw M, Raza K, Kumar K. Selfmanagement strategies used during flares of rheumatoid arthritis in an ethnically diverse population. Musculoskeletal Care 2010;8: Carr A, Hewlett S, Hughes R et al. Rheumatology outcomes: the patient s perspective. J Rheumatol 2003;30: Bingham CO. Flare and disease worsening in rheumatoid arthritis: time for a definition. Int J Adv Rheumatol 2009;7: Crosby LJ. Factors which contribute to fatigue associated with rheumatoid-arthritis. J Adv Nurs 1991;16: Kivitz AJ, Espinoza LR, Sherrer YR, Liu-Dumaw M, West CR. A comparison of the efficacy and safety of celecoxib 200 mg and celecoxib 400 mg once daily in treating the signs and symptoms of psoriatic arthritis. Semin Arthritis Rheum 2007;37: Gladman DD, Hing EN, Schentag CT, Cook RJ. Remission in psoriatic arthritis. J Rheumatol 2001;28: Billing E, McKenna SP, Staun M, Lindqvist U. Adaptation of the Psoriatic Arthritis Quality of Life (PsAQoL) instrument for Sweden. Scand J Rheumatol 2010;39: Coates LC, Cook R, Lee KA, Chandran V, Gladman DD. Frequency, predictors, and prognosis of sustained minimal disease activity in an observational psoriatic arthritis cohort. Arthritis Care Res 2010;62: Holloway I, Wheeler S. Qualitative nursing in research. Oxford: Blackwell, Palominos PE, Gaujoux-Viala C, Fautrel B, Dougados M, Gossec L. Clinical outcomes in psoriatic arthritis: a systematic literature review. Arthritis Care Res 2012;64: Stamm TA, Nell V, Mathis M et al. Concepts important to patients with psoriatic arthritis are not adequately covered by standard measures of functioning. Arthritis Rheum 2007;57: McKenna SP, Doward LC, Whalley D et al. Development of the PsAQoL: a quality of life instrument specific to psoriatic arthritis. Ann Rheum Dis 2004;63: Gossec L, de Wit M, Kiltz U et al. A patient-derived and patient-reported outcome measure for assessing psoriatic arthritis: elaboration and preliminary validation of the Psoriatic Arthritis Impact of Disease (PsAID) questionnaire, a 13-country EULAR initiative. Ann Rheum Dis 2014;73: Dandorfer SW, Rech J, Manger B, Schett G, Englbrecht M. Differences in the patient s and the physician s perspective of disease in psoriatic arthritis. Semin Arthritis Rheum 2012;42: Gronning K, Lomundal B, Koksvik HS, Steinsbekk A. Coping with arthritis is experienced as a dynamic balancing process. A qualitative study. Clin Rheumatol 2011;30: Taylor W, Gladman D, Helliwell P et al. Classification criteria for psoriatic arthritis: development of new criteria from a large international study. Arthritis Rheum 2006;54: Braun V, Clarke V. Using thematic analysis in psychology. Qual Res Psychol 2006;3: Flurey CA, Morris M, Richards P, Hughes R, Hewlett S. It s like a juggling act: rheumatoid arthritis patient perspectives on daily life and flare while on current treatment regimes. Rheumatology 2014;53: Helliwell PS, Fitzgerald O, Strand CV, Mease PJ. Composite Measures in Psoriatic Arthritis: a report from the GRAPPA 2009 annual meeting. J Rheumatol 2011;38: Helliwell PS, FitzGerald O, Fransen J et al. The development of candidate composite disease activity and responder indices for psoriatic arthritis (GRACE project). Ann Rheum Dis 2013;72: Gladman DD, Mease PJ, Strand V et al. Consensus on a core set of domains for psoriatic arthritis. J Rheumatol 2007;34: