Development and validation of a questionnaire to assess facilitators and barriers to physical activity for patients with rheumatoid arthritis, axial spondyloarthritis or psoriatic arthritis

Objectives: The objective was to develop and validate a self-administered questionnaire to identify in people with Inammatory arthritis (IA) Facilitators And Barriers to Physical activity (PA): the IFAB questionnaire. Methods: The development of the questionnaire included a systematic review of barriers and facilitators to PA to identify key themes, face validity assessment by 11 experts and cognitive debrieng with 14 patients. The psychometric properties of the questionnaire were assessed by convergent validity (Spearman correlation) against the modied Health Assessment Questionnaire (mHAQ), the Fear-Avoidance Beliefs Questionnaire subscale for PA and the Tampa Scale for Kinesiophobia, internal consistency (Cronbach α) in 63 IA patients with rheumatoid arthritis (RA), axial spondyloarthritis (axSpA) or psoriatic arthritis (PsA). Reliability and feasibility were assessed in 32 IA patients. Results: The questionnaire comprises 10 items: 4 assessing either barriers or facilitators, 3 assessing barriers and 3 assessing facilitators. The items are related to: psychological status (N=6), social support (N=2), disease (N=1), environmental factors (N=1). In the validation study of 63 patients: 26 RA, 24 axSpA, 13 PsA; mean age 52.8 (standard deviation 16.5) years, mean disease duration 12.5 (12.3) years, 53% women. The questionnaire was correlated (r=0.24) with mHAQ. Internal consistency (Cronbach α 0.69) and reliability (interclass coecient 0.79 [95% condence interval 0.59; 0.88]) were satisfactory, as was feasibility (missing data 12%, mean completion time <5 minutes). Conclusion: The questionnaire allows the assessment of barriers and facilitators to PA in patients with IA. This questionnaire may guide targeted interventions to increase levels of PA in these patients.


Introduction
Axial spondyloarthritis (axSpA), rheumatoid arthritis (RA) and psoriatic arthritis (PsA) are the most prevalent in ammatory rheumatic diseases, representing 2% of the general population [1,2]. They share common characteristics such as in ammation, pain and fatigue, systemic manifestations and can potentially lead to structural changes in joint or spine with loss of function [1,3].
According to the World Health Organization (WHO), physical inactivity (lack of physical activity) has been identi ed as the fourth leading risk factor for global mortality [4]. Patients with these 3 in ammatory arthritis (IA) i.e., RA, SpA or PsA, are more prone to physical inactivity than the general population [5]. For example, only a quarter of axSpA patients met the recommended level of physical activity (PA) [5] and PA objectively measured by a tracker revealed that RA patients performed 200 minutes of PA per week less than healthy subjects [6].
Patients with IA derive speci c bene ts from regular PA. These include, in particular, reduction of pain intensity and risk of cardiovascular diseases [7,8]. For example in AxSpA, increased PA leads to improved physical function and reduced fatigue and disease-related activity [9]. Thus, PA is a key component of clinical practice guidelines for the management of these rheumatic conditions [8,10,11].
Increasing PA is a challenge [12]. Lifestyle changes (such as PA levels) should be addressed by a global approach and by taking into account behavioural barriers to increase chances of success [13]. Regarding PA, barriers and facilitators have been identi ed [14]. Barriers appear to be mostly related to psychological status, and facilitators are linked in part to social support [14,15]. Questionnaires can be used to assess barriers and facilitators, which could be useful in developing interventions tailored to the patient's needs, and thus more likely to be successful. Such a questionnaire could be also valuable in the process of implementing physical activity recommendations [16].
For the general population and for people with osteoarthritis, questionnaires have been developed to assess barriers and facilitators to PA [14,15]. However, barriers in IA may be different due to the speci c burden of the disease, including pain and fatigue, but also structural damage possibly leading to joint or spine functional impairment [3].Furthermore, patients with IA may have erroneous beliefs regarding PA [12]. To our knowledge, no questionnaire assessing perceived barriers and facilitators to PA has been validated for patients with IA.
The aim of this work was to elaborate a self-administered questionnaire to identify the perceived barriers and facilitators to PA in people with IA, and to validate the questionnaire in terms of psychometric properties.

Methods
The study was developed according to COnsensus-based Standards for the selection of health Measurement INstruments (COSMIN) and Outcome Measures in Rheumatology (OMERACT) [17,18].
Several phases were performed, to generate, select and score items for the questionnaire, then to assess the psychometric properties of this questionnaire and to nalise the questionnaire and assess its reliability. These steps are shown in Fig. 1 2.2. Phase 2: Identi cation of the questionnaire items First, each main barrier and facilitator from the systematic review was formulated using the patients' verbatim where possible. Items were presented at either barriers, facilitators or either one of those categories. This preliminary wording was then assessed for face validity, and reworded where needed, by 11 experts in rheumatology or physiotherapy. Finally, the preliminary questionnaire was tested by 14 patients with IA through cognitive debrie ng, and the items were reworded if needed [19]. For this, consecutive outpatients were contacted from one tertiary care hospital in France. They were aged above 18, with de nite RA based on the ACR/EULAR classi cation criteria of rheumatoid arthritis [20], or axSpA based on the Assessment of SpondyloArthritis international Society classi cation criteria [21] with no restriction for co-morbidities and were able to read and write in French language. At that step, items were developed simultaneously in English and French and the quality of the cross-cultural validity was tested using the usual translation and back-translation procedure (29). The following steps were carried out using the French version.

Phase 3: Scoring
Based on the cognitive debrie ng, we chose to rst ask for impact of each item on PA (yes/no), then to rate the impact on PA on a numeric scale ranging from 0 (no impact) to 10 (maximal impact). Rating the impact is important since other authors have demonstrated that the presence or absence of an item is not enough to discriminate between active and inactive participants, and that the extent to which the items impact PA should be noted [22]. Items which can be considered as either barriers or facilitators are rated from − 10 to 10, items which are barriers only are rated from − 10 to 0, and items which are facilitators only are rated from 0 to 10. This scoring has been used previously [22]. When an item was indicated as not affecting physical activity, it was scored at 0.

Population
All patients who satis ed the inclusion criteria, seen in outpatient visits from November 2019 to January 2020 were asked to participate. The inclusion criteria were: age above 18; de nite IA con rmed by the rheumatologist: axSpA (referring to the Assessment of SpondyloArthritis international Society classi cation criteria [21]), RA (referring to the international classi cation criteria of RA [20]) or PsA (referring to the ClASsi cation of Psoriatic ARthritis (CASPAR) criteria [23]); agreement to participate and informed consent; ability to read and write in the language of participating country. There were no restrictions for comorbidities; however, bed-ridden patients were excluded.

Instruments administered
The participants completed self-administered questionnaires: the newly-developed IFAB questionnaire, the Modi ed Health Assessment Questionnaire (mHAQ) to assess functional status [24], the Fear-Avoidance Beliefs Questionnaire subscale for PA (FABQ-PA) to assess patients' fear avoidance and beliefs about PA [25], the Tampa Scale for Kinesiophobia (TSK) to assess the subjective rating of kinesiophobia or fear of movement [26] and the Bath Ankylosing Spondylitis Disease Activity Index (BASDAI) to assess disease activity for patients with AxSpA [27] (online supplementary Table 1). In RA, disease activity was assessed by the physician through the Disease Activity Score 28 (DAS28) [28]. Disease duration, level of pain and patient global assessment were also collated. Finally, the steering committee developed two numeric rating scale global questions related to PA as follows: "when I think about the PA I want to do, I feel held back and limited" and "when I think about the PA I want to do, I feel supported and encouraged" Reliability was explored by test-retest one week after the rst completion. The second completion concerned only the IFAB, and was lled in from home.
All questionnaires were administered in French, using the French versions.

Analysis of psychometric properties and choice of the nal IFAB items
Based on the validation study, the steering committee nalized the questionnaire: the objective was to aim for brevity (to enhance feasibility) without losing content validity, leading to possible deletion of some questions. The selection of the most relevant items for the nal IFAB was based on correlation against legacy measures, correlation between each item and the total score (Spearman's Rho coe cient greater than 0.5, with P < 0.05), clinical relevance (modi able items were preferred), and reliability of each item (intraclass correlation coe cient, ICC > 0.5). Construct validity of each item and of several versions of the IFAB questionnaire were assessed against each of the legacy questionnaires. The hypotheses included: for convergent validity, correlation between the IFAB and the individual items and the legacy questionnaires assessing related concepts (mHAQ, FABQ-PA, TSK); and for divergent validity, absence of correlation with BASDAI, DAS28, level of pain, patient global assessment and disease duration. A correlation between the IFAB and the two simple questions related to PA was also expected. Reliability was assessed on the sub-set of patients with stable symptoms since the rst completion.
The distribution of the scores was evaluated for the questionnaire score and items by the mean, standard deviation, range and median. Floor and ceiling effects were investigated for the questionnaire score and items (accepted maximum for both: 15%). Internal consistency and feasibility were also analysed. Respondent acceptability was considered satisfactory for a frequency of missing data < 15% on the retest. Feasibility was also tested by measuring the duration of completion on 10 participants.

Statistical analyses
For assessment of psychometric properties, a sample size of 60 patients was aimed for validity, and 30 patients for reliability, as proposed by COSMIN, without a formal calculation of power [18]. Statistical analyses were performed using R version 3.5.1. Continuous data were presented as mean (standard deviation (SD)). There was no imputation of missing data.  [29]. ICC was based on the two-way random effect model, and coe cients > 0.70 were considered satisfactory. Internal consistency of the questionnaire was determined by Cronbach's α, and ≥ 0.70 was considered as satisfactory [30].

Systematic review of barriers and facilitators to physical activity
Of 89 references, 10 (11.2%) studies were qualitative reports in RA and were analysed: 3 (49 patients) were focus groups and 7 (99 patients) were individual interviews (Online supplementary Table 2). In all, 148 patients participated: weighted mean age 57.9 years (standard deviation, SD 3.6, range of means 50.0-63.5 years), weighted mean disease duration 13.5 years (SD 5.7, range 6.9-21.0). Barriers and facilitators to PA were reported for general PA in 6 studies and for supervised exercises (e.g. exercise program led by physiotherapist) in 4 studies. Overall, 7 categories of barriers (mean 3.4 (SD 1.9) per study) and 8 categories of facilitators (mean 3.7 (SD 2.1) per study) were reported (Table 1). These could be grouped in 4 major themes psychological status, social support, disease, or environmental factors. The 3 most reported categories of barriers were symptoms of RA (8/10 studies), beliefs, fears and loss of motivation (7/10) and lack of support, exercise knowledge, con dence and con ict in advice from healthcare (4/10). The 3 most reported categories of facilitators were group socialization and social support (8/10), positive beliefs, knowledge about bene ts of PA (5/10) and positive emotions and experiences of PA (5/10).

Psychometric properties of the questionnaire Characteristics of participants included in the validation study
The study included 63 patients (26 (41%) RA, 24 (38%) axSpA, 13 (21%) PsA), mean age 52.8 (SD 16.5) years, mean disease duration 12.5 (SD 12.3) years, 53% women (Table 2 and online Supplementary  Fig. 1). Disease activity was moderate (mean DAS28 3.8 (SD 1.1), mean BASDAI 3.1 (SD 1.4)) and 73% received a biological treatment (Table 2).  Final version of the questionnaire Based on the results and the pre-de ned criteria, the decision was taken to reduce the questionnaire from 14 items to 10 items (Table 3 and (online Supplementary Table 3). In the nal questionnaire, 4 items can be considered either as barriers or facilitators, 3 items as barriers only and 3 items as facilitators only (Tables 3 and 4). The score of the nal list of items can vary between − 70 and 70 (Fig. 1). The items are related to: psychological status (N = 6), social support (N = 2), disease (N = 1), or environmental factors (N = 1) ( Table 3). Items which can be considered as either barriers or facilitators are rated from − 10 to 10, items which are barriers only are rated from − 10 to 0, and items which are facilitators only are rated from 0 to 10. The score of the nal questionnaire can vary between − 70 and 70.   The items are detailed in Table 3.
* Missing data in the questionnaire at retest $Floor and ceiling effects are the % of patients with a minimal or maximal value for the item, which re ects the item is assessed as not being a barrier or a facilitator.

Concurrent validity and internal consistency
Main psychometric properties are shown in Table 4 and online Supplementary Table 4. Convergent validity was partly satis ed, with correlation between the IFAB total score and the mHAQ (r=-0.24, p < 0.05), but no correlation with TSK and FABQ-PA. Divergent validity was satis ed (online Supplementary  Table 4). The questionnaire score had a signi cant positive correlation with the single questions related to the feeling of support and encouragement (r = 0.32, p < 0.05) and a signi cant negative correlation with the feeling of limitation (r=-0.29, p < 0.05) as expected. The internal consistency of the nal version of the questionnaire was good (Cronbach α values = 0.69).

Data completeness and score distribution
The proportion of missing values ranged from 0 to 6% for items and was 12% for the nal score at retest (Table 3).
For use in practice, the questionnaire is available online Supplementary Table 5 in English, and from the authors in French. We propose that in case of one missing item, that item is replaced by the value 0; in case of 2 or more missing items, the questionnaire cannot be scored.
The distribution of items considered as barrier or facilitator only was not normal (Table 4). Floor and ceiling effect were noted when patients were not affected by a barrier (range, 42-73%) or facilitator (range 24-53%). This re ects the fact that not all barriers/facilitators affect all patients, which was expected.
The global score ranges − 70 to 70: in the validation study, there were no oor/ceiling effects for the global score. No oor or ceiling effect was observed for the global score.
The completion time was < 5 minutes.

Reliability
Of the 48 participants who were asked to complete the questionnaires for the test-retest analysis, 32 were analysed (Online Supplementary Fig. 1). The ICC of the IFAB questionnaire was satisfactory (0.78 [95% con dence interval, 0.59; 0.88]) (Table 4).

Discussion
In this study, we have developed an easy to use questionnaire to assess barriers and facilitators to PA for patients with IA. The proposed IFAB questionnaire has 10 items, with a score from − 70 to 70, higher scores indicating higher levels of facilitation and lower levels of barriers. This questionnaire appears to be feasible, reliable and to have satisfactory internal consistency. This questionnaire may guide targeted interventions to increase PA level of patients with IA.
This study has strengths and weaknesses. The generation of themes was based on a systematic literature review of barriers and facilitators to PA for patients; however the review concerned RA only, due to the more extensive literature in this population. Recently, similar barriers and facilitators have been evidenced in patients with AxSpA [12] and to our knowledge barriers and facilitators to PA have not been studied in patients with PsA. Cognitive debrie ng was applied to improve the understandability of items by patients. Cognitive debrie ng is an important step and generally under-used [19]. The development process of the questionnaire involved a variety of experts, including physiotherapists, which is rarely the case. The assessment of psychometric properties was complete, including internal consistency, construct validity, data completeness and reliability [18]. Many questionnaires applied in rheumatology have not undertaken such a complete psychometric assessment [18]. The sample size of patients to assess the validity of the questionnaire (n = 63) was low but is considered by COSMIN as adequate [18]. The sample size for reliability may be considered as low (n = 32) [18]. However, proper statistical analysis was used (ICC) and reliability was satisfactory [18]. No analysis of differential effect in different diseases was carried out. However, all the 3 IA diseases were represented and levels of pain and functional status were equivalent among IA diseases (data not shown). The absence of correlation between the IFAB and questionnaires assessing fear avoidance/beliefs about PA and kinesiophobia was unexpected. However, the IFAB questionnaire assesses several dimensions and some of these are not related to fears (avoidance or kinesiophobia), such as social support or level of symptoms. We believe the global nature of the IFAB could explain the lack of correlations. Furthermore, construct validity showed some correlation with functional status, which was expected and strengthens the face validity of the IFAB questionnaire.
Other questionnaires have been developed to assess barriers to PA in the general population [14] or other disabled populations such as osteoarthritis [15], mobility impairments [31], stroke population [32] or coronary artery disease [33]. In these questionnaires, the number of items used to assess barriers varied from 11 to 63. Only one recently-published questionnaire (the EPPA questionnaire) assessed facilitators of PA [15]. Seven out of ten barriers and facilitators of the EPPA questionnaire were also assessed in the IFAB questionnaire, indicating an expected overlap. However, the EPPA questionnaire is applicable to osteoarthritis, not IA. When comparing with a theoretical framework of behaviour change developed by Canes in 2012, we observed that the IFAB questionnaire covers 7 out of the 14 possible domains (knowledge, beliefs about the capabilities, beliefs about consequences, reinforcement, intentions, environmental factors and resources and social in uence) [34]. This shows the potential interest of the IFAB questionnaire to examine different aspects of the areas of behaviour change.
PA is highly recommended for the IA population, but not performed su ciently [6]. There's been a growing interest patient reported outcomes; the Food and Drug Administration underlines their importance and encourages their use [35]. This study applied recommended methodology for the IFAB questionnaire development and validation. The current questionnaire can be both used for research and clinical practice as it takes less than 5 minutes to complete and has little missing data. This questionnaire has the potential to identify perceptions towards PA in order to address barriers such as mistaken beliefs, and encourage facilitators. It may be useful to implement such a questionnaire during patient education to increase awareness of barriers and facilitators or in clinical trials evaluating PA level to observe the relationship between perceived facilitators and barriers and PA levels. The question remains on how to interpret IFAB scores. In the present study, the median of the score was 3 (range − 70 to 70), with a rst quartile value (de ning the lower 25% of the group) at -5. It might be interesting to propose a targeted intervention to patients with the lower scores, for example negative scores or scores below − 5.
Cultural environment is important to consider when assessing barriers and facilitators. Different aspects of the IFAB questionnaire, such as social environment and psychological status, can be in uenced by culture and habits [36]. The questionnaire was validated only in France. However, the systematic review performed to develop the questionnaire included studies conducted in various countries. Moreover, experts involved for the face validity came from different countries (France, Norway and England). The questionnaire was developed both in English and French following a validated translation and crosscultural adaptation process [37]. The culture of PA can change from country to country and from year to year [38]. Policies and regulations vary across the world. Through national prevention plans, increasing numbers of people are aware of the recommendations to perform PA [5]. The popularity of a sport evolves over the years as shown nowadays by the growing interest in running or tness. Technology is more and more developed and used to promote and track PA and the image of an athletic body is more and more valued [39]. The physical environment is also being adapted to allow regular PA, e.g. through the installation of biking lanes or recreational programs in parks. In addition, the cost of activities tends to decrease. As a consequence, it is possible that this questionnaire will have to be updated in the future.
In conclusion, this short questionnaire may be useful to assess barriers and facilitators in order to increase PA in IA patients. Further studies should assess the relation between IFAB scores and objective PA, and assess the e cacy of interventions to improve PA, based on the IFAB score. This questionnaire could be used to design intervention with bigger chance of success and to help implement physical activity recommendations.

Declarations
Funding: This work was supported by Eli Lilly France and Sandoz France through unrestricted grants. The funders played no role in the design of the study, data collection and analysis, the decision to publish, or the preparation of the manuscript

Con icts of interest/Competing interests: Thomas Davergne, Rikke H Moe, Bruno Fautrel and Laure
Gossec declare that they have no con ict of interest.
Ethics approval: All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards. The validation study was accepted by the ethics committee (CPP Sud-Est III, France, EudraCT 2019-A01413-54, methodology MR03 for non-interventional study).
Consent to participate: Informed consent was obtained from all individual participants included in the study.
Consent for publication: 'Not applicable' for that section.
Availability of data and material: The datasets generated during and/or analysed during the current study are available from the corresponding author on reasonable request.
Code availability: : The codes use in R software during the current study are available from the corresponding author on reasonable request.   Supplementaryonline.pdf