140 Chapter 5 measures compared to the SF-36v2 and the presence of floor or ceiling effects. Data on the time needed to complete the PROMIS CATs, short forms, fixed scale and SF-36v2 were not available. Previous studies, however, reported that the median time needed to complete seven PROMIS CATs was 10.2 minutes, while participants required a median of 5 minutes to complete the SF-36v2 59,60. These studies confirm our conclusion that the selected PROMIS CATs are slightly less feasible to complete than the SF-36v2. However, PROMIS CATs are more precise compared to the SF-36v2 due to the absence of ceiling effects and the limited occurrence of floor effects. Other possible limitations of this study are related to sample size and generalizability of the results, partly due to the overall survey response rate of 33 percent and possible selection bias. The unequal distribution of patients across the three distinct types of inherited bleeding disorders, the limited information on patient characteristics (e.g., race, ethnicity, or educational level) and the unequal distribution among the collected patient characteristics (i.e., sex, disease severity, age group) limited our ability to determine the feasibility, measurement properties and relevance of the PROMIS measures separately for each type of bleeding disorder and for disease severity. In addition, due to the limited number of included patients with IPFDs or RBDs and the heterogeneity within these two disease populations, we were only able to determine the feasibility, measurement properties, and relevance of the PROMIS measures for patients with VWD. Moreover, most patients had a non-severe bleeding phenotype. The results of this study might therefore not be as representative for patients with a severe bleeding phenotype, patients receiving prophylactic treatment, or patients with IPFDs or RBDs. The rare nature of these bleeding disorders makes them inherently difficult to study. We therefore included the results of patients with IPFDs and RBDs in the Supplementary Methods, so that researchers could potentially use our results to inform future studies or pool results. Clinical implications and future research Our study shows that PROMIS measures are reliable and valid instruments to measure physical function, pain interference, fatigue, anxiety, depression, ability to participate in social roles and activities, satisfaction with social roles and activities, and anger in patients with VWD. In contrast to some domains of the SF-36v2 and the PROMIS fixed scale global health, all PROMIS CATs can be used to draw reliable conclusions on individual patient level and can therefore be used in clinical practice. However, before PROMIS CATs can be implemented
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