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

Research question Is a digital behaviour change intervention feasible and acceptable in supporting self-management in patients with Adrenal Insufficiency?

Background Adrenal 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.

Aim To 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 dissemination Findings 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).