title: Meaningful health outcomes for paediatric neurodisability: Stakeholder prioritisation and appropriateness of patient reported outcome measures.
abstract: BACKGROUND
  Health 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.
  
  
  METHODS
  Relevant 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.
  
  
  RESULTS
  A 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.
  
  
  CONCLUSIONS
  We 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.
authors.affiliation: PenCRU, Child Health Group, University of Exeter Medical School, University of Exeter, South Cloisters, St Luke's Campus, Exeter, EX1 2LU, UK. Christopher.Morris@exeter.ac.uk.|PenCLAHRC, University of Exeter Medical School, University of Exeter, South Cloisters, St Luke's Campus, Exeter, EX1 2LU, UK. Christopher.Morris@exeter.ac.uk.
authors.forename: Christopher
authors.initials: C
authors.lastname: Morris
authors.affiliation: PenCRU, Child Health Group, University of Exeter Medical School, University of Exeter, South Cloisters, St Luke's Campus, Exeter, EX1 2LU, UK. A.Janssens@exeter.ac.uk.|PenCLAHRC, University of Exeter Medical School, University of Exeter, South Cloisters, St Luke's Campus, Exeter, EX1 2LU, UK. A.Janssens@exeter.ac.uk.
authors.forename: Astrid
authors.initials: A
authors.lastname: Janssens
authors.affiliation: PenCRU, Child Health Group, University of Exeter Medical School, University of Exeter, South Cloisters, St Luke's Campus, Exeter, EX1 2LU, UK. V.M.Shilling@sussex.ac.uk.|PenCLAHRC, University of Exeter Medical School, University of Exeter, South Cloisters, St Luke's Campus, Exeter, EX1 2LU, UK. V.M.Shilling@sussex.ac.uk.
authors.forename: Valerie
authors.initials: V
authors.lastname: Shilling
authors.affiliation: Council for Disabled Children, National Children's Bureau, London, UK. AAllard@ncb.org.uk.
authors.forename: Amanda
authors.initials: A
authors.lastname: Allard
authors.affiliation: Council for Disabled Children, National Children's Bureau, London, UK. AFellowes@ncb.org.uk.
authors.forename: Andrew
authors.initials: A
authors.lastname: Fellowes
authors.affiliation: Department of Child Health, Royal Devon and Exeter NHS Foundation Trust, Exeter, UK. Richard.Tomlinson@nhs.net.
authors.forename: Richard
authors.initials: R
authors.lastname: Tomlinson
authors.affiliation: Department of Child Health and Paediatrics, Nottingham University Hospitals NHS Trust, Nottingham, UK. Jane.Williams2@nuh.nhs.uk.
authors.forename: Jane
authors.initials: J
authors.lastname: Williams
authors.affiliation: PenCLAHRC, University of Exeter Medical School, University of Exeter, South Cloisters, St Luke's Campus, Exeter, EX1 2LU, UK. J.Thompson-Coon@exeter.ac.uk.
authors.forename: Jo
authors.initials: J
authors.lastname: Thompson Coon
authors.affiliation: PenCLAHRC, University of Exeter Medical School, University of Exeter, South Cloisters, St Luke's Campus, Exeter, EX1 2LU, UK. morwenna.rogers@exeter.ac.uk.
authors.forename: Morwenna
authors.initials: M
authors.lastname: Rogers
authors.affiliation: Social Policy Research Unit, University of York, York, UK. Bryony.Beresford@york.ac.uk.
authors.forename: Bryony
authors.initials: B
authors.lastname: Beresford
authors.affiliation: PenCLAHRC, University of Exeter Medical School, University of Exeter, South Cloisters, St Luke's Campus, Exeter, EX1 2LU, UK. C.Green@exeter.ac.uk.
authors.forename: Colin
authors.initials: C
authors.lastname: Green
authors.affiliation: Nuffield Department of Population Health, University of, Oxford, UK. Crispin.Jenkinson@dph.ox.ac.uk.
authors.forename: Crispin
authors.initials: C
authors.lastname: Jenkinson
authors.affiliation: Department of Rehabilitation Medicine, University of Leeds, Leeds, UK. A.Tennant@leeds.ac.uk.
authors.forename: Alan
authors.initials: A
authors.lastname: Tennant
authors.affiliation: PenCRU, Child Health Group, University of Exeter Medical School, University of Exeter, South Cloisters, St Luke's Campus, Exeter, EX1 2LU, UK. Stuart.Logan@exeter.ac.uk.|PenCLAHRC, University of Exeter Medical School, University of Exeter, South Cloisters, St Luke's Campus, Exeter, EX1 2LU, UK. Stuart.Logan@exeter.ac.uk.
authors.forename: Stuart
authors.initials: S
authors.lastname: Logan
mesh_terms: D000293:Adolescent; D035843:Biomedical Research / Q000458:organization & administration; D002648:Child; D019988:Children with Disabilities / Q000534:rehabilitation* / Q000706:statistics & numerical data; D005260:Female; D006282:Health Personnel; D006305:Health Status Indicators; D006801:Humans; D007223:Infant; D008297:Male; D065886:Neurodevelopmental Disorders / Q000145:classification* / Q000453:epidemiology / Q000628:therapy*; D010287:Parent-Child Relations; D063868:Patient Outcome Assessment; D010372:Pediatrics / Q000458:organization & administration; D036301:Qualitative Research; D011788:Quality of Life