Table 1.

Characteristics of studies selected for review.

Study	Localization	Number of participants	Comorbidities	Exposures investigated in the study (besides hemophilia)	Participants in HC group	Participants in control group	Carrier diagnosis method	Requirements for control group	Method for inquiry about signs and symptoms
Plug et al. (2006)	Netherlands (all the country)	519	Women treated with tranexamic acid, desmopressin or preparations of coagulation factors prior to medical intervention or with other coagulation disorders (other than hemophilia) were excluded	None	274	245	DNA analysis (haplotype or mutation analysis); pedigree analysis and clotting factor dosage (prior to 1985)	Women with negative test for hemophilia, but with a history of familial hemophilia	Questionnaire per letter, based on the validated questionnaire of Srámek et al.21
Siboni et al. (2009)	Italy (patients from Hemophilia and Thrombosis Center, Milan)	228	Conditions or diseases that could explain menorrhagia, use of oral contraceptives or intrauterine device, some treatment used to control menorrhagia or causes of menorrhagia (fibroma, endometriosis, leiomyoma etc.) were registered but did not exclude the patients; women with prophylaxis for bleeding were excluded	Von WIllebrand disease and RBD	31	114	Unknown	Volunteers, friends of patients, of comparable social and cultural context, in good health and without diagnosis of any coagulopathy	Interview with a questionnaire of Srámek et al.21 modified by Podda et al.23
Shahbazi et al. (2012)	Iran (patients from Iranian Comprehensive Hemophilia Care Center, Tehran)	300	All participants should have had at least one full-term pregnancy	Von WIllebrand disease and RBD	47	200	Diagnostic criteria based on international guidelines (not mentioned) under the supervision of hematologists	Women referred by state health services and without coagulation problems referred to in routine consultations, vaccination, family planning etc.	Interview based on MCMDM-1 for DVW22 and Axelsen et al.,24 as well as review of medical records
Olsson et al. (2014)	Sweden (patients from three hemophilia treatment centers)	216	Some patients were using estrogenic oral contraceptive (5) or hormone replacement (3), and no patient was pregnant or less than 3 months of postpartum	None	126	90	DNA analysis (with sequencing of F8 and F9)	Women who are friends of patients, with no history of personal or family bleeding, who had never been referred to specialist service because of bleeding	Interview based on MCMDM-1 questionnaire for DVW22