The impact of severe osteogenesis imperfecta on the lives of young patients and their parents - a qualitative analysis.
BACKGROUND
Osteogenesis imperfecta (OI) is a rare genetic disorder that causes increased bone fragility. Living with, caring for, and parenting a child with OI are all highly demanding and challenging. This study is a temporal analysis of the impact of severe OI on the lives of young patients and their parents.


METHODS
This study was carried out at the Shriners Hospital for Children, a pediatric orthopedic hospital located in Montreal, Canada. Using qualitative interpretative description, we conducted semi-structured interviews with 24 subjects - 12 young patients diagnosed with severe OI and 12 of their parents. The interview data were subject to a predominantly inductive open thematic analysis and a temporal comparative analysis. We did a retrospective chart review to complement our data collection.


RESULTS
We found that the impact of severe OI on the young patients and their parents was characterized by four themes: 1) Starting at the time of diagnosis, a series of stages shaped life and the return to every day "normal", 2) Living with OI was full of "ups and downs" throughout life, 3) Every day "normal" life with OI consisted of significant changes for parents and challenges for the whole family, and 4) Living with OI generated some positive experiences.


CONCLUSIONS
This study contributes to a better theoretical understanding of the impact of severe OI on families. It also has some practical implications for the development of effective support systems for patients with severe OI and their families.
Shriners Hospital for Children, 1529 Cedar Avenue, Montreal, QC H3G 1A6, Canada. joyce.maman.dogba@mail.mcgill.ca.
Maman Joyce
MJ
Dogba
Christophe
C
Bedos
Michaela
M
Durigova
Kathleen
K
Montpetit
Trudy
T
Wong
Francis H
FH
Glorieux
Frank
F
Rauch
D000223:Adaptation, Psychological; D000293:Adolescent; D000328:Adult; D005260:Female; D006801:Humans; D008297:Male; D008875:Middle Aged; D010013:Osteogenesis Imperfecta / Q000523:psychology*; D010290:Parents / Q000523:psychology*; D036301:Qualitative Research; D011787:Quality of Health Care; D013997:Time Factors; D055815:Young Adult