Metabolic Bone Disease Team, Sheffield Children's NHS Foundation Trust, Western Bank, Sheffield S10 2TH, UK.
Health Qual Life Outcomes. 2014 Apr 16;12:54. doi: 10.1186/1477-7525-12-54.
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.
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.
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.
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.
成骨不全症(OI)是一种严重程度不同的疾病,会影响儿童及其家庭的身体、社交和情感福祉。目前尚无关于 OI 患者如何看待其生活质量(QoL)的证据。本研究的主要目的是确定 OI 如何影响儿童及其家庭的生活质量和幸福感。这是为 OI 儿童开发特定疾病生活质量衡量标准的更大项目的第一阶段。
采用目的性抽样法覆盖 OI 人群的多样性。对 10 名儿童(n=10)、10 名家长(n=10)和 5 名医疗保健专业人员进行了 25 次定性访谈。访谈进行了数字录音并逐字转录。确定了有意义的主题、提取和组织主题,并进行了框架分析。
确定了六个主要主题:安全小心、功能降低、疼痛、恐惧、孤立、独立。三组受访者之间有很大的一致性,但父母和孩子之间在独立和恐惧这两个主题上存在差异。
该数据代表为 OI 儿童开发特定疾病生活质量衡量标准的第一步。发现的几个主题与其他生活质量衡量标准相似,但添加了安全小心,特别是与骨折有关,这表明需要为 OI 儿童开发特定疾病的衡量标准。