title: Shaping and managing the course of a child's disease: parental experiences with osteogenesis imperfecta.
abstract: BACKGROUND
  Osteogenesis imperfecta (OI), a rare genetic disease, causes increased bone fragility. The course of childhood chronic conditions particularly rare genetic diseases can be modified by both child and parents. However with limited research, shaping and managing that process is not well understood.
  
  
  OBJECTIVE
  Here we examine how parents of children with severe and mild OI have shaped and managed the condition of their child over time. Our goal is to provide a in-depth understanding of parental responses to OI.
  
  
  METHODS
  This study was carried out in a pediatric orthopedic hospital located in Montreal, Canada. Using the principles of interpretative description, we conducted semi-structured interviews with 48 parents of children diagnosed with OI.
  
  
  RESULTS
  We found that parental responses to their child' disease are constituted by their feelings and their actions. These responses changed over time. We can report four successive phases: an initial reaction, acceptance, normalization and passing the baton. Each stage affected subsequent stages. Every stage was influenced by the severity of OI, parents' individual characteristics, their day-to-day experiences and the entourage.
  
  
  CONCLUSIONS
  Our study contributes to increased understanding of parental responses to OI and to improved parental responses and ultimately the child's coping.
authors.affiliation: Shriners Hospital for Children, 1529 Cedar Avenue, Montreal, QC, Canada H3G 1A6. Electronic address: joyce.maman.dogba@mail.mcgill.ca.
authors.forename: Maman Joyce
authors.initials: MJ
authors.lastname: Dogba
authors.affiliation: Shriners Hospital for Children, 1529 Cedar Avenue, Montreal, QC, Canada H3G 1A6.
authors.forename: Frank
authors.initials: F
authors.lastname: Rauch
authors.affiliation: Center of Laval University Research Hospital 10, rue de l'Espinay, QC, Canada G1L 3L5.
authors.forename: Ghislaine
authors.initials: G
authors.lastname: Tre
authors.affiliation: Shriners Hospital for Children, 1529 Cedar Avenue, Montreal, QC, Canada H3G 1A6.
authors.forename: Francis H
authors.initials: FH
authors.lastname: Glorieux
authors.affiliation: Faculty of Dentistry, McGill University, 3550 University Street, Montreal, QC, Canada H3A 2A7.
authors.forename: Christophe
authors.initials: C
authors.lastname: Bedos
mesh_terms: D000223:Adaptation, Psychological; D000328:Adult; D002170:Canada; D002648:Child; D002908:Chronic Disease; D006233:Persons with Disabilities; D004644:Emotions; D005260:Female; D006776:Hospitals, Pediatric; D006801:Humans; D008297:Male; D009985:Orthopedics; D010013:Osteogenesis Imperfecta; D010290:Parents / Q000523:psychology; D012720:Severity of Illness Index
keywords: Children; Genetics; Illness and rare disease; North America; Osteogenesis imperfecta; Parental responses; Psychosocial issues; Qualitative; Research