title: Quality of life in children and adolescents with Osteogenesis Imperfecta: a qualitative interview based study.
abstract: BACKGROUND
  Osteogenesis Imperfecta (OI) is a disease with varying severity affecting physical, social and emotional well-being of the child and their family. There is no existing evidence on how the OI population regard their quality of life (QoL). The main aim of this study was to determine how OI impacts on the quality of life and well-being of children and their family. It is the first stage of a larger project to develop a disease specific quality of life measure for children with OI.
  
  
  METHODS
  Purposive sampling was used to cover the diversity of the OI population. Twenty-five qualitative interviews were undertaken with children (n = 10), parents (n = 10) and health professionals (n = 5). Interviews were digitally recorded and transcribed verbatim. Significant themes were identified, extracted and organised, undergoing framework analysis.
  
  
  RESULTS
  Six main themes were identified; being safe and careful, reduced function, pain, fear, isolation, independence. There was a large amount of agreement between the three groups of interviewees, although discrepancies did occur between parents and children, with regard to the themes independence and fear.
  
  
  CONCLUSIONS
  This data presents the first step in developing items for a disease specific QoL measure for children with OI. Several of the themes uncovered showed similarity to other QoL measures, but the addition of being safe and careful, particularly in relation to fractures, demonstrated the need for a disease specific measure for children with OI.
authors.affiliation: Metabolic Bone Disease Team, Sheffield Children's NHS Foundation Trust, Western Bank, Sheffield S10 2TH, UK. claire.wagstaff16@btinternet.com.
authors.forename: Claire L
authors.initials: CL
authors.lastname: Hill
authors.forename: Wendy O
authors.initials: WO
authors.lastname: Baird
authors.forename: Stephen J
authors.initials: SJ
authors.lastname: Walters
mesh_terms: D000203:Activities of Daily Living / Q000523:psychology; D000293:Adolescent; D000328:Adult; D002648:Child; D005239:Fear / Q000523:psychology; D005260:Female; D006801:Humans; D057187:Independent Living / Q000523:psychology; D007407:Interviews as Topic; D008297:Male; D008875:Middle Aged; D010013:Osteogenesis Imperfecta / Q000523:psychology*; D010146:Pain / Q000523:psychology; D010290:Parents / Q000523:psychology; D036301:Qualitative Research; D011788:Quality of Life / Q000523:psychology*; D012934:Social Isolation / Q000523:psychology
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