54 Chapter 2 However, one should be careful that in diseases that only have a small effect on HRQoL such a generic instrument might not be sensitive enough to measure a clinically meaningful effect. In that case, a disease specific measurement may still be necessary which need to be developed in patients with autosomal inherited bleeding disorders. Furthermore, it is important that future studies focus specifically on woman due to their hemostatic challenges and the association between bleeding scores and PROs. Finally, with the right PROMs, measuring PROs in patients with autosomal inherited bleeding disorders could help with the –sometimes difficult- decision to start treatment for specific subgroups of patients, for example, patients with a more severe bleeding phenotype, like VWD type 3, who may benefit from prophylactic or other supportive therapy. Keywords Patient reported outcome measures; Quality of Life; Systematic review; Blood coagulation disorders, inherited; von Willebrand disease; Blood coagulation disorders, platelet disorders. Author contributions E.S. van Hoorn en M.E. Houwing designed the study and conducted the search strategy. E.S. van Hoorn and M.E. Houwing screened studies for eligibility and completed the data extraction. E.S. van Hoorn, M.E. Houwing and W. Al Arashi assessed the risk of bias. E.S. van Hoorn, M.E. Houwing and W. Al Arashi interpreted and analyzed the data. E.S. van Hoorn, M.E. Houwing and W. Al Arashi wrote the manuscript. F.W.G. Leebeek, J.A. Hazelzet, S.C. Gouw, R.E.G. Schutgens, S.E.M. Schols, H.F. Lingsma and M.H. Cnossen critically revised the manuscript. H.F. Lingsma en M.H. Cnossen supervised the study. All authors read and approvel the final manuscript. Conflicts of interest F.W.G. Leebeek has received unrestricted research grants from CSL Behring, Shire/Takeda, Sobi and uniQure. He is a consultant for CSL Behring, Shire/ Takeda, Biomarin and uniQure, of which the fees go to the University. He received travel support from Sobi. He is DSMB member of a study sponsored by Roche. S.C. Gouw received unrestricted research grant from Sobi. R.E.G. Schutgens has received unrestricted research and speaker fees from Bayer, Shire, NovoNordisk, Sobi and Octapharma. S.E.M. Schols has received travel grants from Takeda and Bayer, an unrestricted educational grant from Takeda and has served as a steering board member for Roche and Novo Nordisk. M.H. Cnossen has received investigator-initiated research- and travel grants over
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