Amanda Allard

Key health outcomes for children and young people with neurodisability: qualitative research with young people and parents

Objectives To identify key health outcomes, beyond morbidity and mortality, regarded as important in children and young people with neurodisability, and their parents. Design Qualitative research incorporating a thematic analysis of the data supported by the Framework Approach; the International Classification of Functioning, Disability and Health (ICF) provided a theoretical foundation. Setting The study was conducted in community settings. Participants Participants were 54 children and young people with neurodisability: 50 participated in focus groups, and 4 in interviews; 53 parents participated: 47 in focus groups and 6 in interviews. Children/young people and parents were recruited through different networks, and were not related. Results Children/young people and parents viewed health outcomes as inter-related. Achievement in some outcomes appeared valued to the extent that it enabled or supported more valued domains of health. Health outcomes prioritised by both young people and parents were: communication, mobility, pain, self-care, temperament, interpersonal relationships and interactions, community and social life, emotional well-being and gaining independence/future aspirations. Parents also highlighted their childs sleep, behaviour and/or safety. Conclusions Those responsible for health services for children/young people with neurodisability should take account of the aspects of health identified by families. The aspects of health identified in this study provide a basis for selecting appropriate health indicators and outcome measures.

Setting research priorities to improve the health of children and young people with neurodisability: a British Academy of Childhood Disability-James Lind Alliance Research Priority Setting Partnership

Objectives To engage young people, parent carers and clinicians in a systematic process to identify and prioritise research questions regarding ways to improve the health and well-being of children and young people with neurodisability. Design British Academy of Childhood Disability (BACD)-James Lind Alliance research priority setting partnership bringing together patients, carers and clinicians as equal stakeholders. Setting UK health service and community. Methods The BACD Strategic Research Group formed the partnership. A Steering Group was established; charity and professional partner organisations were recruited. Suggestions were gathered in an open survey and from research recommendations for statutory guidance. Items were aggregated to formulate indicative research questions and verified as uncertainties from research evidence. An interim survey was used to rank the questions to shortlist topics. A mixed group of stakeholders discussed the top 25 questions at the final priority setting workshop agreeing a final rank order and the top 10 research priorities. Participants Partner organisations were 13 charities and 8 professional societies. 369 people submitted suggestions (40% non-clinicians). 76 people participated in the interim prioritisation (26 parents, 1 young person, 10 charity representatives, 39 clinicians); 22 took part in the final workshop (3 young people, 7 parents, 3 charity representatives, 9 professionals). Results The top three research priorities related to (1) establishing the optimal frequency and intensity (dose) for mainstream therapies, (2) means for selecting and encouraging use of communication strategies and (3) ways to improve childrens attitudes towards disability. The top 10 included evaluating interventions to promote mobility, self-efficacy, mental health, continence, physical fitness, educational inclusion and reduce impacts of sleep disturbance. Conclusions The methodology provided a systematic and transparent process to identify research priorities that included stakeholders that have typically not contributed to setting the research agenda. The top 10 and other topics identified provide a resource for researchers and agencies that fund research

Meaningful health outcomes for paediatric neurodisability: Stakeholder prioritisation and appropriateness of patient reported outcome measures

BackgroundHealth services are increasingly focused on measuring and monitoring outcomes, particularly those that reflect patients’ priorities. To be meaningful, outcomes measured should be valued by patients and carers, be consistent with what health professionals seek to achieve, and be robust in terms of measurement properties.The aim of this study was (i) to seek a shared vision between families and clinicians regarding key aspects of health as outcomes, beyond mortality and morbidity, for children with neurodisability, and (ii) to appraise which multidimensional patient reported outcome measures (PROMs) could be used to assess salient health domains.MethodsRelevant outcomes were identified from (i) qualitative research with children and young people with neurodisability and parent carers, (ii) Delphi survey with health professionals, and (iii) systematic review of PROMs. The International Classification of Functioning Disability and Health provided a common language to code aspects of health. A subset of stakeholders participated in a prioritisation meeting incorporating a Q-sorting task to discuss and rank aspects of health.ResultsA total of 33 pertinent aspects of health were identified. Fifteen stakeholders from the qualitative and Delphi studies participated in the prioritisation meeting: 3 young people, 5 parent carers, and 7 health professionals. Aspects of health that emerged as more important for families and targets for health professionals were: communication, emotional wellbeing, pain, sleep, mobility, self-care, independence, mental health, community and social life, behaviour, toileting and safety. Whilst available PROMs measure many aspects of health in the ICF, no single PROM captures all the key domains prioritised as for children and young people with neurodisability. The paucity of scales for assessing communication was notable.ConclusionsWe propose a core suite of key outcome domains for children with neurodisability that could be used in evaluative research, audit and as health service performance indicators. Future work could appraise domain-specific PROMs for these aspects of health; a single measure assessing the key aspects of health that could be applied across paediatric neurodisability remains to be developed.

Towards a shared vision for measureable and meaningful health outcomes for children and young people with neurodisability: qualitative research, Delphi survey, systematic review, and stakeholder prioritisation

Towards a shared vision for measureable and meaningful health outcomes for children and young people with neurodisability: qualitative research, Delphi survey, systematic review, and stakeholder prioritisation Christopher Morris, Astrid Janssens, Amanda Allard, Joanne Thompson Coon, Valerie Shilling, Richard Tomlinson, Jane Williams, Andrew Fellowes, Morwenna Rogers, Karen Allen, Bryony Beresford, Colin Green, Crispin Jenkinson, Alan Tennant, Stuart Logan