title: Assessment of the feasibility and clinical value of further research to evaluate the management options for children with Down syndrome and otitis media with effusion: a feasibility study.
abstract: BACKGROUND
  There is clinical uncertainty of the benefits and costs of different treatment options for children with Down syndrome who have glue ear. This study was designed to assess the extent of this lack of knowledge and determine if pursuing further information would be practical, beneficial and cost-effective.
  
  
  OBJECTIVE
  To assess the level and practical effect of current uncertainty around treatment options for children with Down syndrome and glue ear. To assess the feasibility of studying the options for management of glue ear in children with Down syndrome via a randomised controlled trial (RCT) or multicentre prospective cohort study by evaluating the willingness of (1) parents to agree to randomisation for their children and (2) clinicians to recruit participants to a definitive study. To undertake value of information analyses to demonstrate the potential economic benefit from undertaking further research.
  
  
  METHODS
  A feasibility study exploring the views of parents of children with Down syndrome and professionals who have responsibility for the health and education of children with Down syndrome, on the participation in, and value of, future research into interventions for glue ear. Data were collected from parents via self-completed questionnaires, face-to-face interviews and focus groups and from professionals via online questionnaires and a Delphi review exercise. Development of economic models to represent clinical pathways of care and a RCT informed a value of information (VOI) analysis.
  
  
  METHODS
  UK (professionals); East Midlands region of the UK (parents).
  
  
  METHODS
  Parents of children aged 1-11 years with Down syndrome (n = 156). Professionals including audiologists, ear, nose and throat surgeons, audiological physicians, speech and language therapists, and teachers of the deaf (n = 128).
  
  
  METHODS
  Quantitative and qualitative data on parental views and experiences of glue ear and its effects; interventions and treatment received; taking part in research and factors that would encourage or discourage participation; and the importance of various outcome domains to them and for their children. For professionals: information on caseloads; approaches to clinical management; opinions on frequency and significance of the consequences of glue ear for this population; importance of different outcome measures; opinions of interventions and their role in future research; views on health research; facilitators and barriers to recruitment, and participation in research involving RCTs.
  
  
  RESULTS
  The complexity of the experience and individual characteristics of children with Down syndrome poses challenges for the design of any future research but these challenges were not considered by professionals to raise sufficient barriers to prevent it being undertaken. Parents were generally supportive of the need for, and value of, research but identified practical and emotional issues that would need addressing. Glue ear was considered to impact more on speech, language and communication than on hearing. Outcome measures for future research would need to evaluate these elements but measures should be designed specifically for the population. Parents and professionals identified randomisation as a significant barrier to participation. The VOI analyses identified lack of data as problematic but concluded that a future trial involving surgical intervention would be feasible at costs of < £650,000.
  
  
  CONCLUSIONS
  Future research into the benefits of interventions for glue ear in children with Down syndrome would be feasible and could be cost-effective but should be carefully designed to facilitate and maximise participation from parents and professionals responsible for recruitment.
  
  
  BACKGROUND
  The National Institute for Health Research Health Technology Assessment programme.
authors.affiliation: National Institute for Health Research, Nottingham Hearing Biomedical Research Unit, Nottingham, UK.
authors.forename: Heather
authors.initials: H
authors.lastname: Fortnum
authors.affiliation: School of Medicine, University of Nottingham, Nottingham, UK.
authors.forename: Paul
authors.initials: P
authors.lastname: Leighton
authors.affiliation: School of Pharmacy, University of Nottingham, Nottingham, UK.
authors.forename: Murray D
authors.initials: MD
authors.lastname: Smith
authors.affiliation: National Institute for Health Research, Nottingham Hearing Biomedical Research Unit, Nottingham, UK.
authors.forename: Lisa
authors.initials: L
authors.lastname: Brown
authors.affiliation: School of Medicine, University of Nottingham, Nottingham, UK.
authors.forename: Matthew
authors.initials: M
authors.lastname: Jones
authors.affiliation: Nottingham University Hospitals NHS Trust, Nottingham, UK.
authors.forename: Claire
authors.initials: C
authors.lastname: Benton
authors.affiliation: Nottingham University Hospitals NHS Trust, Nottingham, UK.
authors.forename: Elizabeth
authors.initials: E
authors.lastname: Marder
authors.affiliation: Nottingham University Hospitals NHS Trust, Nottingham, UK.
authors.forename: Andrew
authors.initials: A
authors.lastname: Marshall
authors.affiliation: Nottinghamshire Healthcare, County Health Partnerships, Nottingham, UK.
authors.forename: Kate
authors.initials: K
authors.lastname: Sutton
mesh_terms: D000328:Adult; D001291:Attitude of Health Personnel; D002648:Child; D002675:Child, Preschool; D015331:Cohort Studies; D003147:Communication Disorders / Q000191:economics / Q000209:etiology / Q000517:prevention & control / Q000628:therapy*; D003362:Cost-Benefit Analysis; D003697:Delphi Technique; D004314:Down Syndrome / Q000150:complications* / Q000191:economics; D004424:Ear Canal / Q000002:abnormalities; D004739:England; D005240:Feasibility Studies; D005260:Female; D006310:Hearing Aids / Q000191:economics / Q000523:psychology / Q000706:statistics & numerical data*; D034381:Hearing Loss / Q000150:complications / Q000191:economics / Q000209:etiology / Q000628:therapy*; D006801:Humans; D007223:Infant; D007407:Interviews as Topic; D008297:Male; D008875:Middle Aged; D008876:Middle Ear Ventilation / Q000009:adverse effects / Q000191:economics / Q000706:statistics & numerical data*; D018803:Models, Economic; D010034:Otitis Media with Effusion / Q000150:complications / Q000191:economics / Q000628:therapy*; D010043:Outcome and Process Assessment, Health Care / Q000191:economics; D010290:Parents / Q000523:psychology*; D036301:Qualitative Research; D019057:Quality-Adjusted Life Years; D016032:Randomized Controlled Trials as Topic / Q000191:economics / Q000523:psychology* / Q000706:statistics & numerical data; D012107:Research Design; D011795:Surveys and Questionnaires; D055815:Young Adult