Title: Development and randomised feasibility trial of a theory-based digital behaviour change intervention to support self-management for people with Adrenal Insufficiency: the “Support-AI” study
Funder: Health Education England

Abstract: Research question Is a digital behaviour change intervention feasible and acceptable in supporting self-management in patients with Adrenal Insufficiency? BackgroundAdrenal insufficiency (AI) results from cortisol deficiency, a hormone essential for life, caused by failure of the adrenal glands or conditions affecting the hypothalamic-pituitary-adrenal axis. Patients with AI require lifelong glucocorticoid replacement. They need to increase glucocorticoids during illness or major stress and require parenteral hydrocortisone in the event of an Adrenal Crisis (AC), mainly precipitated by infections, to prevent hospitalisation and death; these are called 'sick day rules' [4, 5, 7, 8]. Approximately, 1 in 100 patients die and 1 in 12 are hospitalised at least once/year from AC [9, 10]. Almost 4,000 AC-related hospital admissions, at an estimated annual cost of 6 million, are recorded every year in the NHS for the 25,000 patients with AI in the UK [11]; half of these could be prevented with improved self-management. Patients with AI have impaired quality of life and high morbidity and mortality [12-14]. A systematic review by the applicant (PROSPERO-CRD42018087632) identified self-management barriers relating to nonadherence to glucocorticoids and 'sick day rules' and other behaviours that aid prevention and treatment of AC, such as AC-symptom awareness, having extra tablets, carrying a medical-alert ID and injection kit, and ability to self-inject. Evidence suggests that existing education programmes are ineffective at improving adherence [15] or reducing AC-related hospitalisations [9, 16]. Therefore, a theory-based self-management intervention focused on behaviour change is needed for patients with AI. AimTo develop a digital self-management behaviour change intervention for patients with AI, and to evaluate its feasibility and acceptability in a randomised feasibility trial. Methods The study, underpinned by the MRC Framework [17], the Behaviour Change Wheel (BCW) [18] and the Person-Based Approach [19], will be conducted in two phases over three years.Phase 1 aims to develop a digital intervention to support self-management for patients with AI.1a) Informed by the systematic review and ongoing research conducted by the applicant, four focus groups of patients with AI will elicit potential strategies to overcome barriers and enhance enablers to self-management.1b) BCW methods will be followed to identify potential salient behaviour change techniques and modes of digital delivery. These will be discussed with an Advisory Intervention Panel (10-15 patients, family members, clinicians), using Nominal Group Technique to gain consensus on which should form part of the intervention. A Person-Based Approach using iterative 'think-aloud' technique will guide the development of the website.Phase 2 will test the feasibility, acceptability, and progression criteria of the intervention. A randomised feasibility trial will recruit 80 patients with AI from a university hospital and two patient advocacy groups. Outcome measures will inform a large-scale definitive RCT. Impact and disseminationFindings will be disseminated to patients and health professionals via scientific/patient conferences, social media, and peer-reviewed publications. The 5-year projected benefit includes cost-effective NHS care (reduced hospitalisations and demand for specialist services) and improved outcomes for patients with AI (QoL, adherence to treatment, reduced hospitalisation, and death from AC).

Lay abstract: BackgroundAdrenal insufficiency (AI) results from damage to the adrenals (Primary AI) or the pituitary gland (Secondary AI) leading to cortisol deficiency. Patients with AI require lifelong treatment with medications called glucocorticoids, usually hydrocortisone tablets taken 2-3 times/daily, and need to increase their dose during illness or major stress. They require an injection of hydrocortisone immediately in the event of an Adrenal Crisis (AC) to avoid hospitalisation and potential death. These are called 'sick day rules'. AC is a life-threatening complication mainly triggered by infections, diarrhoea, vomiting or surgery. AI has a significant impact on the patients and NHS services. Patients with AI have impaired quality of life and health problems such as diabetes, obesity, and osteoporosis, often caused by over-treatment with glucocorticoids. Approximately 1 in 100 patients die and 1 in 12 are hospitalised at least once a year from an AC. Almost 4,000 AC-related emergency hospital admissions occur every year in the 25,000 patients living with AI in the UK, which is costing the NHS more than 6 million/year. Half of AC-related hospitalisations could be prevented and complications from glucocorticoid treatment could be minimised with effective self-management. This includes improved adherence to daily glucocorticoids and 'sick day rules', and adoption of behaviours that aid prevention and treatment of AC, including AC-symptom awareness, having extra tablets, carrying a medical-alert ID and injection kit, and self-injection ability. A review of the literature in a systematic way by the applicant showed that current patient education programmes in AI improve patients' knowledge of their treatment. However, they are not effective at improving patients' adherence to 'sick day rules' and preventing AC-related hospitalisations and death. There is therefore a need to adopt a broader self-management approach that is based on theory, focusing on behaviour change and an intervention that is more easily accessible by patients. Aims To develop and test a website-based self-management intervention which aims to support patients to improve adherence to glucocorticoids and 'sick day rules', and adopt behaviours that aid the prevention and treatment of AC. Design and methodsThe research study, guided by behaviour change theories, will be conducted in 2 phases over three years: Phase 1 aims to develop the website-based intervention informed by the earlier systematic review, and an ongoing national survey currently conducted by the applicant and her team. Four focus groups with patients with AI will be conducted to elicit potential strategies for the website intervention to support self-management. Results will be discussed with an Advisory Intervention Panel comprising of 10-15 patients and clinicians to agree on which behaviour change techniques and modes of delivery will inform the website development. Phase 2 aims to test the feasibility and acceptability of the website-based intervention by patients with AI, and to decide if the intervention can be tested for effectiveness in a large-scale randomised controlled trial. Eighty patients with AI, recruited from a university hospital and two patient charities (Pituitary Foundation and Addison's Disease Self-Help Group), will be randomly allocated to the intervention or usual care and followed for 9 months. Patient and Public Involvement (PPI) The applicant has been working with the Study PPI Group comprising of 5 patients since 2016 to develop and refine study design and to support ongoing research. They will continue to advise the applicant alongside her mentors in scheduled meetings at each study milestone. Dissemination and impactResults will be shared with patients and healthcare professional via scientific/patient conferences, social media, websites of collaborating patient charities, and articles written in co-authorship with PPI members. The 5-year projected benefit includes improved health outcomes for patients and cost-effective care for the NHS.

Research activity codes: 7.1 INDIVIDUAL CARE NEEDS
Health categories: METABOLIC AND ENDOCRINE