140 most closely align with the domains that most affect patients with OI. Developing disease-specific Patient-Reported Outcome Measures (PROMS) to assess and follow quality of life is of importance to test the effectiveness of interventions and treatment protocols. There is a wide variation in measurement methods that examine quality of life, which sometimes limits inter-comparison with studies in the literature. Recently, an international interdisciplinary working group reached consensus on a set of PROM tools for people with OI 89. This set will enable OI healthcare teams and systems to compare and to improve their care pathways and quality of care worldwide. Further studies are needed to implement interventions that can be evaluated by this standardized outcome set. Already, European networks and registries are being established to create larger databases. Conduct in-depth analysis into specific sub-areas of quality of life in OI Chapters 2 and 3 objectify different aspects of quality of life. However, the reasons why certain aspects of quality of life are less affected, or not affected (mental health) have not been investigated. There are several theories available that could explain possible outcomes. For example, joint laxity leading to fatigue 90, and deformities affecting physical functioning 91. It is important to explore which symptoms are responsible for the low perceived general health, and in what situations people with OI become fatigued in and why. Observational research can be used to look for causal explanations. In addition, qualitative research can be done to further explore causal relationships and assess patients’ experience and needs. People with OI form an extremely heterogeneous group. This due not only to the different types of OI, which may vary in clinical presentation and associated symptoms, but also because of the different age groups and social circumstances, which may explain differences in quality of life. The use of Chapters 2 and 3 as baseline measurements of different aspects of quality of life may inspire follow-up studies in specific patient groups. Future research into the determinants of quality of life and fatigue is important in order to find interventions that will improve quality of life. Develop treatment protocols for improving quality of life in OI One of the findings in Chapter 2 is that patients with OI experience more pain than the control population. The fact that pain is a concrete and relevant issue that affects all different subgroups can be used to encourage researchers and clinicians to pay better attention to it during follow-up, in research, and in the consulting room. The results also provide evidence and justification for research into treatment of pain. Hypothetically, it could be that some of the pain symptoms are caused by inadequate analgesia. Given the sometimes different body surface in some types of OI, what doses of analgesia should be used in OI is a topic that needs further research. The impor-
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