106 questionnaire such as the ISTH-BAT can also be of benefit in pregnant woman with OI, especially since administration of tranexamic acid to women with post-partum haemorrhage reduces deaths due to bleeding by nearly one third, with no evidence of any adverse effects or complications 56. Strengths and limitations We carried out the largest nationwide study ever performed in adult patients with OI focused on bleeding assessment. With this study a lot of valuable information was obtained which gave a unique insight in the features of bleeding tendency in OI. The results of this study provide a substantial contribution to our knowledge on the symptoms of bleeding disorders in patients with OI. In addition, increased awareness of bleeding complications among health care providers involved in the care of patients with OI may contribute to improved quality of life in these patients. Albeit the Self-BAT is a validated questionnaire for different mild bleeding disorders 15,16, it is not specially developed for OI. Due to the explorative design of this cohort study and the absence of a matched control group, no relative risk estimates could be calculated. Also the use of an self-reporting BAT has some limitations because symptoms reported by patients may be influenced by the fact that several patients are already familiar with the potential relation between OI and an increased bleeding tendence. Furthermore, recall of symptoms and treatments can be difficult, especially when the presence of bleeding symptoms spans a lifetime and might be sporadic due to a low amount of haemostatic challenges. This is also claimed by Moenen et al, who also emphasises that although an elevated BAT score increases the patients likelihood of having an mild bleeding disorder, a specific disorder cannot be diagnosed based on the BAT 57,58. The main value of the BAT is well established in the assessment of a bleeding disorder and provides a structured, complete diagnostic anamneses. Abnormalities can justify extensive laboratory testing because a distinctive bleeding history is a prerequisite for the diagnosis of any bleeding disorder 12,59. We used the BAT for an descriptive purpose to gain more insight in the bleeding tendency in OI. For this reason we also described the different domains of the Self-BAT, which has been done earlier 60. Conclusions The abundant manifestations of bleeding and bruising described in this large cohort suggests that a mild bleeding disorder should be considered in patients with OI. Despite some limitations of a self-report questionnaire, there are very likely more bleeding problems in OI compared to the normal population. Since bleedings due to surgery, tooth extraction, menstruation and obstetrics are more common and can have major clinical consequences, proactive therapy might be considered
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