A Mixed Methods Approach to Implementing the Canadian Spinal Bulbar Muscular Atrophy Guideline

BACKGROUND: Spinal bulbar muscular atrophy (SBMA) is an X-linked neuromuscular disease that disproportionately affects Indigenous people in Canada. The first Canadian guidelines for the management of SBMA were recently published. There is limited guidance from the literature to inform optimal implementation strategies. METHODS: A mixed methods approach was used to determine SBMA guideline implementation strategies. A combination of activities (surveys, roundtable discussion, modified Delphi consensus exercise with experts, Indigenous engagement) was employed to gather opinions from multiple stakeholders and experts (physicians, allied healthcare providers, people with living experience [PWLE] including Indigenous people and patient organizations). RESULTS: Barriers to guideline implementation included lack of awareness of the disease and the need for comprehensive multidisciplinary teams to address the multisystem symptoms and complications of SBMA. A survey of current practice patterns demonstrated several areas for improvement, including attention to cardiac symptoms, laryngospasm and care for female carriers. Roundtable discussion with health experts and patient advocacy groups centered on the following themes: (1) awareness, (2) resources, (3) knowledge translation and (4) policy support. The Indigenous engagement process with the community guiding circle emphasized the need for ongoing involvement of PWLE, Indigenous communities, leaders and health networks in the implementation process. The modified Delphi process with physician experts led to the development of specific implementation strategies, with emphasis on education for healthcare providers and PWLE, including the development of written materials. CONCLUSION: Intentional partnerships with PWLE, Indigenous communities, patient organizations and healthcare professionals are essential to meaningful guideline implementation.