41 Chapter 2 QoL, which is why specifically designed questionnaires focused on determining factors of QoL in adults with OI are essential to improving QoL and are currently being developed. Identifying specific outcomes that are associated with improved or decreased QoL in OI is important to guide timing and nature of interventions and to design research aimed at optimizing well-being of adults with OI 21,30. For example, Dahan-Oliel and colleagues 21 performed a systemic review of previously mentioned studies 14–17 and concluded that for both children and adults with OI pain, scoliosis, activity limitations, and participation restrictions caused by decreased limited function are associated with lower levels of physical QoL and need to be addressed to promote QoL. When interventions are planned, a follow-up measurement of QoL can indicate the effect of these interventions on the different health domains and as such, the impact of these interventions can be measured. In our adult OI service we have tried to identify factors that might positively influence the QoL in people with OI. For this purpose, a value-based health care program has been developed to identify factors that we can influence in our service and that are measurable by QoL questionnaires. These aspects would be consistently monitored through the years. A very important influence on the development of a value-based health care program is the input of the OI group regarding what they consider important for their QoL. Limitations and future plan This study reports on baseline measurements of the QoL in 322 adult patients with OI measured by the SF-36 questionnaire. The SF-36 is a generalized, QoL questionnaire that is not specific for people with OI. This makes the data susceptible to temporary biases such as a recent fracture. However, the SF-36 is wellvalidated and widely used; therefore, it is a valid tool to evaluate QoL for patients with OI. As mentioned earlier, the development of OI-specific questionnaires is important and in progress; the results of this study can serve as basis for their development. We compared our patient data against the data of reference populations collected more than two decades ago. Nonetheless, the reference populations were unique and representative of the Dutch population. The SF-36 is sensitive to fluctuations in health 4, which makes it suitable to measure QoL over a longer period or before and after a procedure. As such, we will aim to present a longitudinal overview of QoL in patients with OI through measurements of QoL and its influences at different time points.
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