126 Chapter 5 Adaptive Tests (CAT) 25. With CAT, after the first question, subsequent questions offered to the patient are based on previous answers 26. The use of PROMIS CATs therefore reduces questionnaire burden, while providing a more tailored and reliable measurement in comparison to existing PROMs 17,25,27-29. Previous research has shown that a selection of PROMIS measures, including fixed scales and short forms derived from the item banks, perform well in children and adults with hemophilia 17,21,22. However, it is unclear if PROMIS measures are suitable for patients with VWD, IPFDs and RBDs. Due to the X-linked inheritance pattern, the hemophilia population predominantly consist of men. Moreover, hemophilia patients frequently receive prophylactic treatment to prevent (spontaneous) bleeding. The majority of patients with VWD, IPFDs and RBDs, however, are women and their presentation, diagnosis and management differ significantly from that of men with hemophilia 9. This study aims to evaluate the feasibility, measurement properties and relevance of nine PROMIS measures for adults with VWD, IPFDs, and RBDs. METHODS Study design, participants, and data collection This cross-sectional study included participants from three previously performed nationwide cross-sectional studies on VWD, IPFDs and RBDs in the Netherlands. These are the Thrombocytopathy in the Netherlands (TiN) study, and the Rare Bleeding Disorders in the Netherlands (RBiN) study, and the von Willebrand in the Netherlands – Prospective (Win-Pro) study. The TiN study was performed between 2016 and 2018, and the RBiN study between 2017 and 2019. The WiN-Pro study inclusions were performed from 2019 to 2022. These three studies included patients from all six hemophilia treatment centers in the Netherlands. The inclusion criteria for the WiN Pro study were similar to those of the WiN study and the exact inclusion criteria have been published elsewhere 3,9,12,30. For this specific study, we approached adult Dutch-speaking patients that previously participated in either the WiN-Pro, TiN or RBiN study and gave permission to be contacted for follow-up studies. The exact inclusion criteria for this study differed per type of bleeding disorder and can be found in the supplementary information. In summary, patients with severe or moderate VWD were included if they participated for two years in the WiN-Pro study and received treatment at the hemophilia treatment center Erasmus University
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