Shannon van Hoorn

141 PROMIS in VWD, IPFDs and RBDs 5 in research and routine care additional research should be performed on the feasibility of the PROMIS CATs. First, the number of PROMIS CAT items administrated is higher compared to the SF-36v2. Patients with no problems or disease symptoms have to answer a maximum of 12 questions to reach the CAT stopping rule. Adjustments of this stopping rule should be evaluated to explore if it is possible to reduce questionnaire burden 61. Second, other aspects of feasibility such as the comprehensibility of the PROMIS CATs and SF-36v2 in patients with low health literacy and the cost for administration should be evaluated. Thirdly, additional research should be performed to evaluate the feasibility and measurement properties of the PROMIS measures in patients with IPFDs and RBDs. The results of patients with IPFDs and RBDs provide an indication that the PROMIS measures might also be feasible, reliable, and valid in these patient populations. However, additional research should be performed for confirmation. Lastly, not all PROMIS measures might be relevant for patients with VWD, IPFDs and RBDs. Previous studies on important PROs for this patient population primarily identified bleeding symptoms which are not included in either the PROMIS measures or the SF-36v2 62. Further research should be performed to determine the best set of PROMIS CATs or short forms in combination with a bleeding disorder specific questionnaire to enable measurement of all relevant PROs in this patient population. Preferably, (inter)national consensus should be achieved on this standard set of PROMs to facilitate the comparison between patient population, the general population and other healthcare settings 23. CONCLUSION The studied PROMIS CATs, short form anger and fixed scale global health are a valid and reliable alternative for the SF-36v2 in patients with primarily nonsevere forms of VWD. Moreover, the PROMIS CATs and short form anger can be used to assess the health-related quality of life of this patient population on both group and individual patient level and can therefore be used in clinical practice. The PROMIS fixed scale global health can be applied to compare the health-related quality of life of this patient population on group level. Further research is necessary to evaluate the feasibility and measurement properties of the PROMIS measures in adults with IPFDs and RBDs. Moreover, additional research should be performed to reduce the completion burden of the PROMIS CATs and to determine the optimal combination of generic PROMIS CATs with disease-specific questionnaires to measure PROs in patients with VWD, IPFDs, and RBDs.

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