Feasibility and impact of a physical activity and lifestyle program for Aboriginal families with Machado-Joseph disease living in the Top End of Australia

Brief Name: Feasibility and impact of a physical activity and lifestyle program for Aboriginal families with Machado-Joseph disease living in the Top End of Australia Why: Physical activity and lifestyle programs are scarce for people with hereditary ataxias and neurodegenerative diseases. Aboriginal families in the Top End of Australia, who have lived with MJD for generations, co-designed a physical activity and lifestyle program, called the ‘Staying Strong Toolbox’. The aim of this study was to explore the feasibility and impact of the program on walking and moving around. What: Participants were informed of study requirements in their preferred language by local community researchers and provided either written or oral consent. Each participant communicated in their preferred language throughout the study. Staying Strong Toolbox Program The program was tailored to each individual through use of the Toolbox workbook (Carr 2021). Based on the Staying Strong framework, the workbook has six domains for keeping yourself strong on the inside and outside: ‘Exercising your body’, ‘going country’, ‘searching for good medicine’, ‘keeping yourself happy’, ‘families helping each other’, and ‘something important to do’ (Carr 2019) Within each of the six domains, the workbook includes a range of activities (tools in the Toolbox). The participant selects activities of their choice from each domain to be included in their individualised program, according to their functional level: Strong (independently mobile), Wobbly (mobilising using a walking aid), Wheelchair (‘wheelchair’ dependent) (Carr 2021). Groups of activities within each domain of the workbook and the workbook prototype has been published elsewhere (Carr 2021). Using the workbook, participants (either alone, with family or physiotherapist (JC) and community researchers) selected from the range of activities within each domain, by ticking activities they wished to do to keep walking and moving around. Individuals discussed activities they had selected with the physiotherapist (JC), who provided guidance where necessary. This guidance included advice on how to tailor tasks to the participant’s functional level to ensure the most physical benefit could be gained, and to determine whether tasks could be performed independently or would require assistance. Lastly, participants set a specific goal to work on. Their goal and chosen activities were collated into a weekly planner by the physiotherapist (JC) in collaboration with each participant, to form a four-week program. Duration for set up process of each program ranged between 1-2 hours. Participants carried out their program over a four-week period. Delivery • Each participant had a copy of their weekly plan (on paper or as image on phone). • Each participant undertook their Toolbox activities as per their weekly planner with flexibility to change. • For each activity on each occasion, participants identified the assistance they required and who they required assistance from (e.g. alone, family members and/or the physiotherapist (JC), and was provided accordingly. • Participant performance of activities was informally assessed by the physiotherapist (JC) throughout the program and guidance provided as required. • The weekly planner was revised with participants weekly by the physiotherapist (JC) to establish whether participants felt activities needed to be included or removed. Discussions ranged between 30 minutes and 2 hours. • On program completion, participants were provided with a printout of their own home program based on activities they had done throughout the program pilot. Activities • Minimum activity target for each participant was to be physically active for at least one hour, three times a week, over the four-week program period at a ‘somewhat hard’ level of exertion (RPE 12-14). Facilitators • The physiotherapist (JC) in collaboration with local community researchers provided: o Set up and assistance with each participants’ program in each community. o In-person assistance throughout each four-week program period as required. o Coaching and encouragement throughout the program. o Daily check-ins on weekdays via phone with participants in regard to their progress, unless participants made first contact. Transport • Transport requirements were determined by participants, provided either by family, MJD Foundation support workers, or researchers. • Vehicles were provided by the MJD Foundation with shared access. Vehicles included a four-wheel drive vehicle (4WD) and wheelchair accessible bus on Groote Eylandt and a 4WD (three weeks) and car (one week) in Ngukurr. Equipment available • Equipment was available from the MJD Foundation: exercise bike, fishing gear, camp cooking gear, balls, water carriers, stationery, portable printer, EPIRB, 4WD recovery gear, fuel, basketball, football, stopwatch. • Light snacks and refreshments were provided when researchers accompanied participants (e.g. cold drinks, fruit and/or sandwiches/bread/damper). Who Provided: Research Team Aboriginal community researchers including community Elders (GL, GlL, JC, BD, OD) and physiotherapists with extensive experience in rural and remote setting (JC). How: Programs were conducted face to face with JC and community researcher partners or with community research partners alone. Some participants also trained independently as well as independently accompanied by family members. Where: See above in ‘What’ section - ‘Location’ When and how much: See above in ‘What’ section: ‘Delivery’ and ‘Activities’ Tailoring: See above in ‘What’ section - Staying Strong Toolbox program’ Modifications: Participant performance of activities was informally assessed by the physiotherapist (JC) throughout the program and guidance provided as required. The weekly planner was revised with participants weekly by the physiotherapist (JC) to establish whether participants felt activities needed to be included or removed. Discussions ranged from 30 minutes to 2 hours. How well: Adherence with the program was assessed (reach > 80% of the minimum activity target) for each participant. A research diary was used to collect adherence data. Activities were self-reported by participants and/or observed by the research team according to participant preference. Activity data collected was as follows: activities completed, locations, assistance required, supports, duration, repetitions, attendance and reasons for non-attendance where relevant, serious adverse events or coinciding community events. References: Carr JJ, Lalara J, Lalara G, O'Hare G, Massey L, Kenny N, et al. 'Staying strong on the inside and outside' to keep walking and moving around: Perspectives from Aboriginal people with Machado Joseph Disease and their families from the Groote Eylandt Archipelago, Australia. PLoS One. 2019;14(3). Carr JJ, Lalara J, Lalara G, Lalara G, Daniels B, Clough AR, et al. Staying Strong Toolbox: Co-design of a physical activity and lifestyle program for Aboriginal families with Machado-Joseph disease in the Top End of Australia. PLoS One. 2021;16(2):e0244311.