Osteogenesis Imperfecta Federation Europe, Heffen, Belgium.
Osteogenesis Imperfecta Foundation, Gaithersburg, MD, USA.
BMC Public Health. 2024 Nov 28;24(1):3318. doi: 10.1186/s12889-024-20555-0.
The IMPACT Survey explored the humanistic, clinical, and economic burden of osteogenesis imperfecta (OI) on individuals with OI, their families, caregivers, and wider society. Two previous publications report research methodology, initial insights of the survey, and cost of illness of OI. Here, we present data on the impact of OI on the quality of life (QoL) of adults with OI and explore potential drivers of this impact.
The IMPACT Survey was an international mixed methods online survey in eight languages (fielded July-September 2021), aimed at adults (aged ≥ 18 years) or adolescents (aged 12-17 years) with OI, caregivers (with or without OI) of individuals with OI, and other close relatives. Survey domains included demographics, socioeconomic factors, clinical characteristics, treatment patterns, QoL, and health economics. We conducted a descriptive analysis of the QoL data, as well as exploratory regression analyses to identify drivers of impact of OI on QoL (independent associations between patient characteristics and the impact on QoL).
1,440 adults with OI participated in the survey. The proportion who reported an impact of OI on their QoL across individual areas in the physical, socioeconomic, and mental well-being domains ranged between 49 and 84%. For instance, 84% of adults reported an impact of OI on the types of leisure activities they could do and 74% on the type of job they could do. More severe self-reported OI and higher fracture frequency were consistently identified as drivers of OI's impact on QoL. The proportion of adults who reported worrying about different aspects of their lives due to their OI, such as mobility loss, future fractures, and ageing, ranged between 31 and 97%.
IMPACT provides insights into the humanistic burden of OI on adults, revealing that OI has a substantial impact on the QoL of adults. OI severity and fracture frequency were consistently identified as drivers of impact on QoL across all domains. Understanding these drivers may aid in identifying areas for targeted interventions, such as fracture prevention.
IMPACT 调查探讨了成骨不全症（OI）对 OI 患者、其家人、照顾者和更广泛的社会的人文、临床和经济负担。之前有两篇出版物报告了调查的研究方法、初步结果和 OI 的疾病成本。在这里，我们介绍了 OI 对 OI 成年患者生活质量（QoL）的影响的数据，并探讨了这种影响的潜在驱动因素。
IMPACT 调查是一项国际性的混合方法在线调查，使用了八种语言（2021 年 7 月至 9 月进行），旨在针对 OI 患者（年龄≥18 岁）或青少年（年龄 12-17 岁）、OI 患者的照顾者（有无 OI）以及其他近亲。调查领域包括人口统计学、社会经济因素、临床特征、治疗模式、QoL 和健康经济学。我们对 QoL 数据进行了描述性分析，并进行了探索性回归分析，以确定 OI 对 QoL 影响的驱动因素（患者特征与 QoL 影响之间的独立关联）。
1440 名 OI 成年患者参与了调查。报告 OI 在身体、社会经济和心理健康领域的个人领域对其 QoL 有影响的比例在 49%至 84%之间。例如，84%的成年人报告 OI 影响了他们可以从事的休闲活动类型，74%影响了他们可以从事的工作类型。自我报告的 OI 更严重和更高的骨折频率始终被确定为 OI 对 QoL 影响的驱动因素。报告因 OI 而担心生活的不同方面，如行动能力丧失、未来骨折和衰老的成年人比例在 31%至 97%之间。
IMPACT 提供了关于 OI 对成年人的人文负担的见解，表明 OI 对成年人的 QoL 有重大影响。OI 严重程度和骨折频率始终被确定为所有领域 QoL 影响的驱动因素。了解这些驱动因素可能有助于确定针对骨折预防等领域的目标干预措施。
