title: Positive parental attitudes to participating in research involving newborn screened infants with CF.
abstract: BACKGROUND
  Information regarding recruitment of infants to research studies following the diagnosis of cystic fibrosis (CF) via newborn screening (NBS) is not currently available. This study aimed to assess parental attitudes and the feasibility of recruiting and retaining both NBS infants with CF and healthy control infants to a longitudinal, observational study.
  
  
  METHODS
  All infants underwent pulmonary function tests (PFTs) at ~3 and ~12months of age. Infants with CF had additional combined chest high resolution computed tomography (HRCT), bronchoscopy and broncho-alveolar lavage (BAL) at ~12months of age. Parental attitude questionnaires (PAQs) were administered to all parents following the ~3month PFTs and to parents of infants with CF after completion of all tests at ~12months.
  
  
  RESULTS
  86% (92/107) of families whose infant had CF consented to participate, of whom 92% had PFTs at ~3months of age with 99% of these having PFTs at ~12months of age. Recruitment of healthy controls was feasible but more challenging; 29% of those contacted agreed to participate; 73% of these had PFTs at ~3months of age; of whom 83% had repeated PFTs at ~12months of age. Completed PAQs were received from 71% of parents, (both of CF and healthy infants) at ~3months and from 58% parents of infants with CF at ~12months. Responses from the PAQs were generally positive, 95% of parents indicated they would recommend participation in such studies to other families. Discrepancies between responses at 3 and 12months suggested that parental understanding of what the research entailed developed during the course of the study.
  
  
  CONCLUSIONS
  The high recruitment and retention rates for newly diagnosed CF NBS infants to this observational study are encouraging. These findings will help inform future study design both in the field of CF and other conditions diagnosed by NBS.
authors.affiliation: Great Ormond Street Hospital for Children NHS Foundation Trust, London, WC1N 3JH, UK. jane.chudleigh@kcl.ac.uk
authors.forename: Jane
authors.initials: J
authors.lastname: Chudleigh
authors.forename: Ah-Fong
authors.initials: AF
authors.lastname: Hoo
authors.forename: Deeba
authors.initials: D
authors.lastname: Ahmed
authors.forename: Ammani
authors.initials: A
authors.lastname: Prasad
authors.forename: Denise
authors.initials: D
authors.lastname: Sheehan
authors.forename: Jackie
authors.initials: J
authors.lastname: Francis
authors.forename: Sarah
authors.initials: S
authors.lastname: Buckingham
authors.forename: Jacqui
authors.initials: J
authors.lastname: Cowlard
authors.forename: Lena
authors.initials: L
authors.lastname: Thia
authors.forename: The Thanh Diem
authors.initials: TT
authors.lastname: Nguyen
authors.forename: Janet
authors.initials: J
authors.lastname: Stocks
mesh_terms: D001290:Attitude; D035843:Biomedical Research; D003256:Community Participation; D003550:Cystic Fibrosis / Q000175:diagnosis*; D005240:Feasibility Studies; D006801:Humans; D007223:Infant; D007231:Infant, Newborn; D015997:Neonatal Screening; D010290:Parents / Q000523:psychology*; D018579:Patient Selection; D011795:Surveys and Questionnaires