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Family experience with osteogenesis imperfecta type 1: the most distressing situations

2018-09Journal article Restricted access
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dc.contributor.authorSantos, Margarida Custódio dos
dc.contributor.authorPires, Ana Filipa
dc.contributor.authorSoares, Kelly
dc.contributor.authorBarros, Luísa
dc.date.accessioned2017-08-21T15:13:36Z
dc.date.available2017-08-21T15:13:36Z
dc.date.issued2018-09
dc.description.abstractBackground: Osteogenesis imperfecta (OI) is a rare genetic disorder characterized by decreased bone mass and increased bone fragility. Despite increasing research on the biomedical aspects of the disease, only a few studies focus on the psychosocial implications of living with OI. This study aimed to explore the situations that are perceived by OI type-1 children, their parents, and siblings, as being the most distressing and stressful in their experience with the disease. Methods: Seven families of children diagnosed with OI type 1 for longer than four years participated. An in-depth semi-structured interview with open-ended questions was used to separately collect each participant’s (mother, father, patient, and sibling) subjective report of their experience. Interviews were audiotaped and a qualitative discourse analysis was performed. Results: Pain and fractures, hospitalization, home recovery, back to school and time of diagnosis emerged as the most challenging situations. Time of diagnosis was only mentioned by parents. Some commonalities but also relevant differences in the subjective experience of the same situations, depending on the family role, were found. Conclusions: Our results reinforce the assumption that OI is a family matter and point to the importance of providing comprehensive and family-centered health and educational services tailored to each family member and to the different situations faced by these families.pt_PT
dc.description.versioninfo:eu-repo/semantics/publishedVersionpt_PT
dc.identifier.citationSantos M, Pires AF, Soares K, Barros L. Family experience with osteogenesis imperfecta type 1: the most distressing situations. Disabil Rehabil. 2018;40(19):2281-7.pt_PT
dc.identifier.doi10.1080/09638288.2017.1334236pt_PT
dc.identifier.urihttp://hdl.handle.net/10400.21/7338
dc.language.isoengpt_PT
dc.peerreviewedyespt_PT
dc.publisherTaylor & Francispt_PT
dc.relation.publisherversionhttp://www.tandfonline.com/doi/abs/10.1080/09638288.2017.1334236?journalCode=idre20pt_PT
dc.rights.urihttp://creativecommons.org/licenses/by-nc-nd/4.0/pt_PT
dc.subjectOsteogenesis imperfectapt_PT
dc.subjectFamilypt_PT
dc.subjectStressful situationpt_PT
dc.subjectSubjective experiencept_PT
dc.subjectRare diseasept_PT
dc.titleFamily experience with osteogenesis imperfecta type 1: the most distressing situationspt_PT
dc.typejournal article
dspace.entity.typePublication
oaire.citation.endPage2287pt_PT
oaire.citation.issue19pt_PT
oaire.citation.startPage2281pt_PT
oaire.citation.titleDisability and Rehabilitationpt_PT
oaire.citation.volume40pt_PT
rcaap.rightsrestrictedAccesspt_PT
rcaap.typearticlept_PT

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