Title: Development and feasibility of a mobile health behaviour change intervention to prevent adrenal crisis in patients with primary adrenal insufficiency- The Prevent Study
Funder: Health Education England

Abstract: Background PAI is a disorder in which the adrenal glands are destroyed by an autoimmune process resulting in hypoadrenalism, this requires patients to take lifelong GRT, and adjust medication appropriately during acute illness to prevent an AC. AC are preventable if patients follow appropriate steroid management advice. Although education is important in patients with PAI, evidence shows that AC and death still occur in well informed patients who have received prior education. Thereby, there is a need to understand the reasons behind this lack of knowledge application, to develop and test an intervention that prevents AC. Research question Does development of a mobile health intervention aid the self management of GRT in preventing AC? Aims 1.Investigate why patients with PAI fail to apply their knowledge of GRT during acute illness to prevent AC. 2.Identify which behaviour change interventions and modifiable factors are preferable to patients in preventing AC. 3.Test the feasibility of using mHealth to deliver behaviour change intervention to prevent AC Methods Phase1 - Systematic Review Systematic review and meta-analysis will aim to answer the following questions: What types and efficacy of behavioural change interventions prevent AC in patients with PAI and what modifiable factors using the COM-B framework influence self-management of adrenal crises? Findings of the review will be utilised in phase 2. Phase 2- Mixed method study Patients with PAI (n=300) from six hospitals and ADSHG will be invited to participate in a prospective study to identify those who have experienced AC (EAC) and who have averted AC (PAC). Qualitative interviews will then be performed on selection of patients (n=6+ 6) and family (n= 6 +6) looking to answer the following questions: 1. Why do patients with PAI not apply knowledge they have to apply sick days rules when in need? 2. Which behaviour change interventions, techniques and modifiable factors (using the COM-B framework) do patients and family/friends believe are effective and acceptable? Phase 3- Co-Design Workshops Using findings from phase 2, a purposive sample of patients with PAI (n=5) and their family/friends (n=2) will be invited to attend three co-design workshops to develop a mobile health (mHealth) behavioural intervention with co-design group members. Phase 4 - Feasibility Study Using a feasibility RCT study design, patients will be randomised (on a 1:1 basis) to mHealth behavioural intervention and standard care (n=25) or standard care based on current education (n=25). Participants will be followed up for 12 months post randomisation and a selection will be interviewed to assess acceptability and improvements to the mHealth app. Anticipated impact and disseminationThe feasibility study will be performed to test methodology and parameters to inform a future full RCT. The primary outcome of the full RCT will provide evidence of mHealth application and its impact on occurrence of adrenal crisis, risk of death and subsequently reduced associated implications of hospital admission, morbidity, mortality and patient's quality of life. Findings will be shared with participants, patient support groups and health professionals by publishing in peer review journals, oral presentations and online forums.

Lay abstract: Background: Addison's Disease (AD) affects around 8400 people in the UK. AD is a chronic condition resulting from adrenal gland failure, where the body no longer produces sufficient cortisol and associated hormones. This requires the patient to take life-long daily steroid tablets. The adrenals are two small glands that sit on the kidneys, producing hormones which have important functions; regulating the body's metabolism, controlling blood pressure and are essential during acute illness and stressful events. Extra steroid replacement is essential for maintaining the body's responses during illness/infection/surgery. When patients with AD are sick (e.g. diarrhoea/vomiting), injured or before surgery they are unable to make enough cortisol leading to an Adrenal Crisis (AC). AC is a shortage of cortisol. Warning signs include Low blood pressure, Low glucose, Nausea, Vomiting, Dizziness and Impaired consciousness. AC affects around 1 in 12 AD patients. In the last year there were 4,329 episodes due to preventable AC. Having an AC results in patients being too unwell to work, requiring help from family and/or health care professionals, hospital outpatient attendance and/or admission to hospital, and can lead to death. AC are preventable if patients follow guidelines called 'sick day rules' including increasing the patient's regular dose of oral steroid or giving an injection of hydrocortisone. Despite teaching patients about sick day rules, hospital admissions and death still occur. Research conducted by the applicant and others confirms that patients with AD do have the necessary knowledge and can adhere to the required medication on a day-to-day basis. However, in times of illness, they do not adapt their dose of steroid replacement and require additional help from family and/or health care professionals. Therefore we need to further understand the reasons behind this. We need to develop and test interventions that improve adherence to sick day rules, resulting in reduced complications, hospital admissions and deaths. Aims: 1) To investigate why AD patients do not apply their knowledge to adapt their steroid dose during episodes of acute illness and/or stress. 2) To explore interventions of behaviour change with patients/families 3) To develop/test usability and acceptability of a digital mobile health behavioural intervention application (app) to adapt/change the dosage of steroids to prevent an AC. Design & Methods: The study has four phases. The objective of each phase includes: Phase 1: Review of existing evidence identifying research and interventions previously used to prevent ACs. Explore factors aiding a patient's ability to personally manage steroid replacement during acute illness in order to prevent AC. Phase 2: Exploring patients' and family/friends' views and experience of enablers and barriers to adapt/change steroid dose in order to prevent AC. Phase 3: Using interactive workshops involving patients, family/friends, health professionals and digital technology experts to co-design a suitable App. Phase 4: To assess the usability/acceptability of the App versus standard care (education advice). Participants will be followed for 9 months. Patient & Public Involvement: AD patients have been involved in developing research questions, study design and data collection method selection and will continue to advise throughout via the project advisory group. A PPI representative will also be involved in disseminating findings. Dissemination: A short lay language article and summary of the research will be developed with PPI. Findings will be shared, and publicised, via patient support groups and made available online for participants. Results will be shared among health care professionals through publishing in research journals and presentations at national/international meetings and conferences. PPI will contribute and co-author research publications and present study findings at meetings and conferences. Results will be used to inform the design of future randomised controlled trials testing the effectiveness of the intervention.

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