118 Materials and methods Study design An explorative study was undertaken in the Expert Center for adults with OI, Isala Hospital, Zwolle, the Netherlands. The Medical Ethics Committee of the Isala Hospital, Zwolle, the Netherlands, confirmed that the Medical Research Involving Human Subjects Act did not apply (reference number: 200612). All patients with a clinical diagnosis of OI who were invited to take part in this study signed informed consent for participation. Evaluation of bleeding tendency The self-BAT is a self-administered bleeding assessment tool validated in Canada and the Netherlands 12,13 and is based on the International Society on Thrombosis and Haemostasis Bleeding Assessment Tool (ISTH-BAT)14,15. The 14 self-BAT domains cover self-reported events of epistaxis, cutaneous bleeding, minor wounds, haematuria, gastrointestinal bleeds, oral cavity bleeds, prolonged bleeding after trauma, surgeries or tooth extraction, menorrhagia, postpartum haemorrhage, muscle, joint and central nervous system bleeds, and other bleedings. Each domain scores from 0 (absence of bleeding symptoms) to 4 (symptoms requiring extensive medical intervention). The distinction between zero points and 1 point is of critical importance since a score of 1 means that the symptom meets the minimal criteria defining a significant bleeding. This distinction between the different scores is described by the ISTH Scientific and Standardization Committee 16 and was independently determined by two trained researchers (KG and GM). Conflicting scores were reassessed and resolved by consensus. A total bleeding score (range 0-56) was calculated by the sum of scores for all BAT domains. A total bleeding score in men ≥ 4 and in women ≥ 6 was defined by Deforest et al. and Punt et al. as an increased bleeding tendency 12,13. In this study, a total bleeding score of >6 was considered an increased bleeding tendency in women. Study population and selection procedure In total, 328 patients with a clinical diagnosis of OI, according to the classification of van Dijk and Sillence 1 known in our centre, were invited to digitally fill in the self-BAT questionnaire between October 2019 and August 2020 (Figure 1). In total, 195 of 328 (60%) questionnaires were filled in and included in the study 17.
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