Aims:
- Develop a theoretically informed online self-management programme for IBD fatigue, pain and urgency/FI, based on CB principles, our extensive previous work and reviews, input from people with IBD and what is known to work for these symptoms in other conditions (WP1a).
- Develop an online training package for IBD nurses to provide telephone support alongside the online self-management programme (WP1b).
- Conduct a feasibility study to test the intervention developed (WP1c).
Work package 1a: focus groups and individual interviews (informing intervention development)
Aim: To understand how people with IBD experience and self-manage these symptoms and what they wanted from an intervention, to inform the development of the intervention to be tested in the trial.
Methods: Face-to-face and online interviews with 26 people in 5 focus groups and 14 individual interviews, recorded and transcribed verbatim. Analysis by researchers and people with IBD, guided by the Analytical Hierarchy Framework (paper 1) and framework analysis (paper 2). Eight people with IBD assisted in the analysis and interpretation of results. Research Ethics Committee (REC) Approval (REC reference: 17/WA/0349/IRAS number: 228902).
Changes from the original proposal: It rapidly became apparent that our original plan to interview people in separate groups about symptoms of fatigue, pain and urgency/FI was not going to work, as participants could not talk about one without mentioning the others and that there was a strong inter-relationship between symptoms. We therefore talked about the symptoms together in the groups and interviews.
This work was reported in two papers:
Paper 1: Patient strategies for managing the vicious cycle of fatigue, pain and urgency in Inflammatory Bowel Disease: impact, planning and support7
Key findings: Figure 2 shows the three core themes which emerged: The Negative Impact of Symptoms, Positively Taking Control, and Seeking and Receiving Support. Multiple coexisting symptoms had a persistent and stark impact on physical and emotional well-being and necessitated adjustments and limitations in working, social and intimate spheres of life. Negative vicious cycles were created by unpredictable symptoms which are challenging and impact each other. Self-managing symptoms included managing diet, pacing, acceptance of background levels of fatigue, pain and urgency, seeking support, exercising and attending to mental well-being.
FIGURE 2 Living with fatigue, pain and urgency: themes from focus groups and interviews. (Figure not reproduced here; see the original article.)
Relation to other parts of the programme: Fatigue, pain and urgency/FI are troublesome for patients, especially in combination, suggesting that these should be addressed simultaneously. Participants reported several strategies for self-management, providing evidence to inform development of our self-management intervention.
Paper 2: Developing an online program for self-management of fatigue, pain, and urgency in Inflammatory Bowel Disease: patients’ needs and wants8
Key findings: Figure 3 shows the three core themes which emerged. People reported attempting to manage the three symptoms. They recognised the combined influence of food, drink, stress and exercise on all symptoms. They requested an online intervention, available on several platforms, including symptom and medication management, and activity and symptom tracking.
FIGURE 3 Patient coping strategies and preferences for content and functionality of an online self-management intervention for fatigue, pain and urgency/incontinence related to IBD.8 Reproduced with permission from Fawson et al.,8 licence from Springer Nature number 6146040877708. (Figure not reproduced here; see the original article.)
Relation to other parts of the programme: Patients reported numerous ways of self-managing symptoms of fatigue, pain and urgency/FI related to IBD and expressed their needs for content, design and functionality of the proposed intervention.
Additional work not in application: We decided that we also needed to interview IBD nurse specialists as they were intended to be the facilitators for the intervention.
Paper 3: Inflammatory bowel disease nurses’ views on taking on a new role to support an online self-management programme for symptoms of fatigue, pain and urgency: a qualitative study to maximise intervention acceptance9
Some materials within this section have been reproduced with permission from Matthias et al.9 This is an Open Access article distributed in accordance with the terms of the Creative Commons Attribution (CC BY 4.0) licence, which permits others to distribute, remix, adapt and build upon this work, for commercial use, provided the original work is properly cited. See: https://creativecommons.org/licenses/by/4.0/. The text below includes minor additions and formatting changes to the original text.
Aim: We aimed to explore IBD nurses views on our proposed online intervention and their role in facilitation.
Methods: We conducted 5 focus groups with 45 participants. Themes were identified using thematic analysis.
Key findings: We identified four themes (Figure 4):
- Most were positive about undertaking the role, but they were concerned about the time needed to facilitate the programme in an already overloaded clinical workload.
- They wanted training and support if undertaking facilitation and wanted options to contact the research team if a patient crisis arose.
- They were concerned about being able to separate the facilitator role from the IBD nurse role, fearing patients would use facilitation sessions to ask questions about disease management rather than aspects of the intervention.
- They wanted clarity on eligibility criteria and how participants would be recruited.
FIGURE 4 Summary of themes and subthemes from IBD nurse interviews.9 Reproduced with permission from Matthias et al.9 This is an Open Access article distributed in accordance with the terms of the Creative Commons Attribution (CC BY 4.0) licence, which permits others to distribute, remix, adapt and build upon this work, for commercial use, provided the original work is properly cited. See: https://creativecommons.org/licenses/by/4.0/. The figure includes additions and formatting changes to the original text. (Figure not reproduced here; see the original article.)
Relation to other parts of the programme: This interview study with IBD nurses helped to refine the proposed online intervention with a view to sustainable implementation in clinical practice. Refinements included in-depth training of nurses as facilitators, including supervised ‘practice patient’ sessions and assessment of competence as a facilitator, production of a training manual for reference and minimisation of additional workload for nurses through reducing patient contact.
Limitations of WP1a interviews: As with all qualitative work, it is not known how representative the volunteers for interviews are of people living with IBD or of IBD nurses more generally. We had originally intended to interview people about fatigue, pain and urgency separately, but they seemed unable to discuss one without discussing the others, so we decided to cover all three symptoms in each group, which led to richer data emerging.
Relation of WP1a to other parts of the programme: We used the information from these interviews with people with IBD and IBD nurses and our previous literature reviews to develop our online IBD-BOOST intervention and facilitator training package. In particular, we used the expressed needs of people with IBD for content and functionality and took into account the IBD nurses’ concerns about finding the time to support the intervention, changing from the originally proposed four 30-minute telephone calls to one call plus online messaging for communication with participants, when designing IBD-BOOST.
Work package 1a: intervention development. Co-design of an online guided self-management programme
Additional work not in the application: We decided that we needed to conduct a systematic review of associations and interventions for urgency/FI in IBD as we could find none in the literature to inform that part of our intervention
Paper 4: Targets for interventions for faecal incontinence in inflammatory bowel disease: a systematic review10
Aim: To explore the evidence on the associations of urgency/FI in IBD and the effectiveness of interventions to inform the IBD-BOOST intervention content.
Methods: Seven databases were searched in October 2017. We also conducted a manual search of reference lists. Four researchers independently screened references and extracted data.
Key findings: Eighteen studies were included; 14 on associations, 4 on interventions; FI was reported in 12.7–76% of 5924 participants. Disease activity, loose stool, female gender, childbirth, previous surgery, anal sphincter weakness or fatigability, anxiety and depression were associated with FI, but causation could not be inferred. The four intervention studies were small (34 participants in total), uncontrolled and included surgery (sphincter repair and sacral nerve stimulation) and tibial nerve stimulation, each of which improved FI. There were no controlled studies testing interventions for FI in IBD to inform our intervention.
Relation to other parts of the programme: We used the information from this review to plan the content of our online IBD-BOOST intervention. In particular, it told us that nobody had done what we were planning to do before, and which factors we could usefully target with our intervention for urgency/FI.
Intervention development: Some materials within this section have been reproduced with permission from Sweeney et al.11 This is an Open Access article distributed in accordance with the terms of the Creative Commons Attribution (CC BY 4.0) licence, which permits others to distribute, remix, adapt and build upon this work, for commercial use, provided the original work is properly cited. See: https://creativecommons.org/licenses/by/4.0/. The text below includes minor additions and formatting changes to the original text.
We used the data from WP1a, this new review and our previous systematic reviews30,31 to inform the development of an online self-management intervention. CB models of fatigue and pain in conditions, such as multiple sclerosis and rheumatoid arthritis, suggest that disease factors (e.g. inflammation) trigger symptoms, but then an interaction of cognitive, behavioural, emotional and physiological responses to symptoms may strengthen and perpetuate them.32,33 Online delivery of CB interventions for chronic pain and fatigue in other medical conditions appear to be effective, with evidence of enhanced effects through the addition of minimal support from healthcare professionals (HCPs).34–38 Empirical modelling and systematic reviews of risk factors and interventions for IBD-related fatigue,3,30 FI10 and chronic abdominal pain in IBD suggest that similar interventions, addressing emotional, behavioural and/or cognitive responses to these symptoms, might reduce the impact and severity of the symptoms.11
The emerging theoretical model was based on a CB model, which explains how thoughts interact with emotions, behaviour and physical symptoms. Unhelpful thought patterns often become trapped in a vicious cycle. In long-term conditions where physical symptoms are triggered by disease factors, unhelpful thoughts, emotions and behaviour can perpetuate troublesome physical symptoms, such as fatigue, pain and urgency experienced by people with IBD. CB theory-based interventions help people learn alternative ways of thinking, through which they can interrupt this negative interaction, which might help alleviate some of the symptom burden and improve patients’ QoL. An evidence-based logic model based on CB theories was used to define the specific intervention techniques (Figure 5).
FIGURE 5 Logic model developed for the IBD-BOOST intervention. BIPQ, Brief Illness Perception Questionnaire; CBRQ, Cognitive Behavioural Responses to Symptoms Questionnaire; EQ-5D, EuroQol-5 Dimensions; IBD-F, Inflammatory Bowel Disease Fatigue; IBDQ, Inflammatory Bowel Disease Questionnaire; PHQ-9, Patient Health Questionnaire-9 items; SEMCD6, Self-Efficacy for Managing Chronic Disease 6-item Scale; VAS, visual analogue scale; VSI, visceral sensitivity index. Reproduced with permission from Sweeney et al.11 This is an Open Access article distributed in accordance with the terms of the Creative Commons Attribution (CC BY 4.0) licence, which permits others to distribute, remix, adapt and build upon this work, for commercial use, provided the original work is properly cited. See: https://creativecommons.org/licenses/by/4.0/. The figure includes additions and formatting changes to the original text. (Figure not reproduced here; see the original article.)
Our approach was informed by the Medical Research Council (MRC) Framework39 for developing and evaluating complex interventions guidance, which outlines the importance of utilising evidence-based theory and models in the design and development process. We also adopted a ‘person-based approach’,40 which emphasises the value of fully understanding the target population and securing their ongoing involvement in the design process, to ensure that the intervention is appropriate and acceptable. The ‘person-based approach’ was designed for digital interventions, and involvement of end users in the development of web-based interventions in IBD outpatient settings has been shown to reduce barriers to adoption.41,42
Throughout the development process, we consulted with 87 PPI patients with IBD, as well as IBD nurses and other health professionals, whose feedback helped refine the online programme content (e.g. number of sessions and session length) and functionalities (e.g. facilitator messaging), including the use of visual aids, e-mail reminders and graphical tracking of symptoms. They also contributed by helping decide the visual features of the intervention including the name ‘IBD-BOOST’ which was chosen for its positive, encouraging image and the colours, fonts and layout of the web-content for accessibility (Figure 6). Inclusivity was considered throughout, resulting in age and gender-neutral references and illustration characters. Forty-five IBD nurses assisted the process through the focus groups reported above, which informed the delivery model of the facilitator support. They helped identify the potential barriers, particularly regarding the limited resources available, resulting in a reduction of patient-facilitator telephone calls from four to one.
FIGURE 6 The IBD-BOOST navigation page that shows style and colour scheme of the intervention co-designed with IBD patients. (Figure not reproduced here; see the original article.)
The final intervention comprised 12 web-based sessions (8 core and 4 symptom-specific sessions: Appendix 1, Table 8), with one 30-minute facilitator phone call following session 1 and subsequent weekly in-site messaging for 12 weeks. Participants were able to complete their own ‘vicious circle’ of symptoms thoughts and behaviours and during the phone call work with their individual facilitator to understand and modify these. On initial testing, each session took 45–60 minutes to complete, and it was possible to undertake these flexibly, interrupt and rejoin at the same place, with the intended use to be approximately one session per week. As well as text, there were audio recordings and additional optional activities such as symptom diaries. We ensured an accessible reading age and style, with use of unisex cartoons and patient quotes from the interviews reported above (see Appendix 1, Figure 21).
Work package 1b: develop a training package for facilitators to provide intervention support
We developed a remote training programme (four 1-hour sessions plus a practice patient call) for intervention facilitators. A manual with quick reference guides was provided for easy reference.
Work package 1c: feasibility testing of the online intervention
Intervention feasibility testing was undertaken throughout the co-design development process with PPI patients (n = 87). Once the intervention design and production were completed, 31 people with IBD were given 8 weeks access to the whole intervention. They provided feedback through web-based surveys or telephone calls; 97% of PPI patients provided feedback after 1 week, 77% after 4 weeks and 68% at the end of the 8-week period. In addition to the formal feasibility testing feedback, feedback was also provided by participants in-programme following each session on how helpful, relevant, easy to navigate and motivating they rated sessions.
The feasibility testing provided very encouraging support for the intervention programme, particularly referring to the visual aspects which were considered professional, bright and clear and the functionality was described as ‘easy to use’. The post development feasibility testing confirmed the value of this co-design process; sessions were rated as understandable, relevant and having an appropriate tone and length. Setting and reviewing goals related to the content covered in the session were rated as easy to complete and useful. A few aspects needed adjustments.
During initial feasibility testing, the patients worked through the programme independently without any facilitator support: two-thirds felt that being able to contact a facilitator would have been helpful and that they would have been likely to contact them for reassurance, validation and queries relating to the content. Overall, users were motivated to continue and were highly likely to recommend it to a friend with IBD.
Paper 5: A novel digital self-management intervention for symptoms of fatigue, pain and urgency in inflammatory bowel disease: describing the process of development11
This detailed paper described the process of developing the supported digital self-management intervention, enabling others to replicate our processes.
Relation to other parts of the programme: We used theory and integrated stakeholders’ views to inform development of an evidence-based digital intervention for fatigue, pain and urgency in IBD. This is the first online self-management intervention designed to address these multiple symptoms with the aim of improving the QoL and reducing the symptom burden of IBD.
After the initial feasibility testing, we conducted a more formal feasibility study, specifically for pain in IBD (symptom chosen pragmatically as it fitted within the PhD of a student, Sweeney, working with IBD-BOOST).
Paper 6: Pain management in inflammatory bowel disease: feasibility of an online therapist supported CBT-based self-management intervention12
Some materials within this section have been reproduced with permission from Sweeney et al.12 This is an Open Access article distributed in accordance with the terms of the Creative Commons Attribution (CC BY 4.0) licence, which permits others to distribute, remix, adapt and build upon this work, for commercial use, provided the original work is properly cited. See: https://creativecommons.org/licenses/by/4.0/. The text below includes minor additions and formatting changes to the original text.
Aim: To test the feasibility of the IBD-BOOST programme, specifically in people reporting pain in IBD.
Methods: A single-arm pre–post design with nested qualitative interviews was used. Twenty individuals with IBD and chronic pain were recruited through an NHS clinic or online via an IBD charity. Individuals who indicated a pain-interference score of ≥ 4/10 (Brief Pain Inventory) and met inclusion criteria were invited to participate. Outcomes included recruitment and retention rates, pain interference and severity, QoL and psychosocial measures.
Key findings: Of 145 individuals contacted via an NHS clinic, 55 (37.9%) responded. Two individuals were recruited from the online study advertisement. Twenty out of 57 (35.1%) met screening and eligibility criteria. Eighty-five per cent of the sample engaged with intervention sessions, and 55% completed at least 5/9 sessions. Eighty per cent of recruited participants completed the post-intervention questionnaire at week 9. The mean score for overall acceptability was 43.4 (0–70). Qualitative feedback demonstrated the value of thought monitoring and facilitator support. Scores improved for QoL and pain self-efficacy and reduced for depression, anxiety, pain catastrophising and avoidance resting behaviour.
Relation to other parts of the programme: The online IBD-BOOST programme for chronic IBD-related pain appeared to be feasible and acceptable. The study suggested positive effects for improving QoL and reducing psychological distress. We used the information to inform the design of our main trial of the IBD-BOOST intervention. In particular, it reinforced the need for a facilitator to support participants in IBD-BOOST and helped us to choose our outcome measures.
Limitations: The co-design work and feasibility testing were with self-selected volunteers and so may not be representative of people with IBD more generally.
Additional work in WP1 not in the application: We also decided that we needed to develop a measure of costs as we could find none available that was suitable for our RCT.
Paper 7: Development and test–retest reliability of a new, self-report questionnaire assessing healthcare use and personal costs in people with inflammatory bowel disease: the Inflammatory Bowel Disease Resource Use Questionnaire (IBD-RUQ)13
Part of this section is reproduced under licence from BMJ Publishing group (licence number 6146060054282).
Aim: We developed a self-report patient questionnaire to capture key resource utilisation from health services, patient and societal perspectives.
Methods: The Inflammatory Bowel Disease – Resource Use Questionnaire (IBD-RUQ), developed by a multidisciplinary team, including patients, comprises 102 items across the 6 categories of outpatient visits, diagnostics, medication, hospitalisations, employment and out-of-pocket expenses over the past 3 months. The test–retest reliability of the IBD-RUQ was studied by administering it twice among patients with IBD with a 2-week time gap. The intraclass correlation coefficients and the average cost from the healthcare, societal and patient perspectives, between test and retest assessments, overall and by service category, were summarised.
Key findings: The IBD-RUQ captures health service use, employment and out-of-pocket expenses. Of 55 patients who completed the first questionnaire, 48 completed the retest questionnaires and were included in the analyses. Test–retest reliability for categories of medications, diagnostics, specialist outpatient and inpatient services, and days off work due to IBD ranged from moderate to excellent; primary care visits showed more limited reliability. The annualised average self-reported health service, out-of-pocket and loss of productivity costs were £4844, £320 and £545 per patient, respectively. The IBD-RUQ is a reliable and valid self-report measure of resource utilisation in adults with IBD and can be used to measure costs associated with IBD.
Relation to other parts of the programme: We used the IBD-RUQ for Health Economic evaluation in the RCT below. The lead author Chris Roukas also obtained funding from Bowel Research UK to undertake a PhD on the costs of IBD and has conducted a larger survey and further validation and analysis on the costs of IBD to the individual and to the health service. Two other research studies are using the IBD-RUQ.