Shannon van Hoorn

83 Patient-relevant health outcomes: A delphi study 3 some participants completed the first Delphi survey in February, while others completed the same survey in November. This delay between the first and second Delphi round may have led to a slightly lower response rate on the second and subsequently third Delphi round. The experienced difficulties during the recruitment of patients, caregivers and healthcare professionals might also have repercussions for the results of this study. While patients with all types of autosomal inherited bleeding disorders were approached and encouraged to participate in this study, the patients and caregivers panel mainly includes patients with Von Willebrand disease. The results of this panel might be less representative for patients with an inherited platelet function disorder or rare bleeding disorder. The rare nature of these diseases makes them inherently difficult to study. Implications for research and practice The identification of which health outcomes are important for patients with autosomal inherited bleeding disorders is the first steps towards valuebased healthcare implementation in this patient population. Health services aiming to implement value-based healthcare can use our results to guide their decisions on which health outcomes should be measured in clinical care to assess and improve patient value for patients with inherited bleeding disorders. Additional validation, however, in both a national and international setting is recommended to ensure that the health outcomes identified in this study are also seen as important by a wider group of patients with autosomal inherited bleeding disorders, their caregivers and healthcare professionals. To assess patient value in reliable manner across bleeding disorders, populations, research and clinical practice further operationalization of the identified health outcomes is crucial. Subsequently, it is essential to reach consensus on a methodology or set of measurement tools to assess the identified important health outcomes for patients with autosomal inherited bleeding disorders. Future studies are needed to reach consensus on which measurement instruments best capture the value of the identified health outcomes and, in the case of non-existing disease specific instruments, to create a suitable, valid and reliable measurement instrument. Furthermore, additional research could focus on synthesizing these study results and the various comparable studies performed in patients with hemophilia into one overarching recommended set of important health outcomes for all types of bleeding disorders. This would create uniformity and

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