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. 2013 Sep 30:13:153.
doi: 10.1186/1471-2431-13-153.

The impact of severe osteogenesis imperfecta on the lives of young patients and their parents - a qualitative analysis

Maman Joyce Dogba  1 , Christophe Bedos, Michaela Durigova, Kathleen Montpetit, Trudy Wong, Francis H Glorieux, Frank Rauch
Affiliations

The impact of severe osteogenesis imperfecta on the lives of young patients and their parents - a qualitative analysis

Maman Joyce Dogba et al. BMC Pediatr. .

Abstract

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.

Conclusion: 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.

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Figures

Figure 1
Figure 1
The impact of severe OI on parents and young patients.
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References

    1. Kazak AE, Reber M, Snitzer L. Childhood chronic disease and family functionning- a study of phenylketonuria. Pediatrics. 1988;81:224–230. - PubMed
    1. van den Tweel XW, Hatzmann J, Ensink E, van der Lee JH, Peters M, Fijnvandraat K, Grootenhuis M. Quality of life of female caregivers of children with sickle cell disease: a survey. Haematologica. 2008;93:588–593. - PubMed
    1. Welch V, Hatton C, Emerson E, Robertson J, Collins M, Langer S, Wells E. Do short break and respite services for families with a disabled child in England make a difference to siblings? A qualitative analysis of sibling and parent responses. Children Youth Serv Rev. 2012;34:451–459.
    1. Zurynski Y, Frith K, Leonard H, Elliott E. Rare childhood diseases: how should we respond? Arch Dis Child. 2008;93:1071–1074. - PubMed
    1. Forlino A, Cabral WA, Barnes AM, Marini JC. New perspectives on osteogenesis imperfecta. Nat Rev Endocrinol. 2011;7:540–557. - PMC - PubMed

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