Management of choking in Motor Neuron Disease: patient and public involvement in a co-produced mixed-methods study protocol

BACKGROUND: Up to 70% of people living with Motor Neuron Disease (plwMND) report distressing episodes of choking (a feeling of suffocation or airways blockage) due to involvement of bulbar muscles. There is an evidence gap on management of choking in plwMND, which is mirrored by the absence of guidance and training for healthcare professionals. Choking in plwMND is associated with other signs of bulbar involvement, such as communication difficulties due to dysarthria, which can be a barrier to effective participation in research. In this study protocol, we describe an innovative approach to meaningful involvement of plwMND, to co-produce a research project on management of choking tailored to their needs and aligning with their priorities. METHODS: Phase 1 (Patient and public involvement co-design meeting): We first engaged in a meeting with plwMND, carers and healthcare professionals, to identify their priorities in relation to improving management of choking in MND. To ensure inclusive participation, we used the Theory of Change framework; people with communication difficulties were supported by a Speech and Language Therapist through their chosen augmentative and alternative communication aids. A key priority identified was the need to produce evidence-based guidance for management of choking tailored to the needs of plwMND. Phase 2 (Healthcare research study protocol on management of choking): based on the outcome of Phase 1, a multidisciplinary team (including individuals with lived experience) designed a mixed-methods, multi-centre research project based on four work packages (WPs). WP1: systematic review of the existing scientific literature on management of choking; WP2: interviews and focus groups with people with lived experience of choking in MND; WP3: co-design of a clinical algorithm detailing interventions to manage choking, based on WP1 and 2; WP4: algorithm development and dissemination with continuous involvement and input from people with lived experience through a Patient and Public Involvement co-applicant and patients' representatives. DISCUSSION: In plwMND and choking, barriers to inclusion in PPI and research range from reduced accessibility due to physical disabilities, to discriminatory sampling due to communication issues. Through inclusive engagement at the outset, plwMND highlighted a mismatch between their priorities (gaining knowledge about choking and feeling empowered to manage it at home) and available evidence (lack of guidance and training for healthcare professionals). This research project aligns with what matters to most affected patients, their carers and healthcare professionals, aiming to fill an evidence gap through new guidance to manage choking tailored to their needs.