Natural history & quality of life in Glanzmann thrombasthenia & Bernard Soulier syndrome: An observational study from India

格朗茨曼血小板无力症和伯纳德-苏利埃综合征的自然病程和生活质量:一项来自印度的观察性研究

阅读:1

Abstract

Background & objectives Inherited platelet function disorders (IPFDs) are not well studied as compared to haemophilia and other bleeding disorders. Present study is aimed to understand the natural history and quality-of-life (QoL) in the two well studied IPFDs i.e. Glanzmann thrombasthenia (GT) and Bernard Soulier syndrome (BSS). Methods This is an ambispective, observational study. Demographics, medical data, mortality due to bleeding, comorbidities and treatment products were recorded. Health related quality of life (HRQoL) was captured using EuroQol five-dimensional questionnaire (EQ-5D), 36-Item short form health survey (SF-36) and functional assessment of chronic illness therapy (FACIT) scales. The severity of bleeding was assessed by annual bleed rate (ABR) and International Society on Thrombosis and Haemostasis - Bleeding assessment tool (ISTH-BAT) score. Results The median (interquartile range; IQR) age of 76 study participants (64 GT, 12 BSS) was 14 yr (9-19 yr). Epistaxis, ecchymosis, gingival bleed, gastrointestinal bleed, and soft tissue bleed were the commonest clinical manifestations. Menorrhagia was seen in all females in the reproductive age group. There was a statistically significant difference in the mean ISTH-BAT scores between GT and BSS (P=0.016). Platelet transfusion was the main mode of treatment; none of the patients in the present series were on activated recombinant factor VII (rFVIIa) therapy. Between 2000 and 2025, 13 deaths were reported (due to bleeding) mainly due to inaccessibility to treatment or treatment products. The relationship between quality of life (QoL) scores and ISTH-BAT score was weak. Interpretation & conclusions The need for optimal treatment strategies to improve QoL and providing timely access to specific treatment products to prevent mortality is underscored.

特别声明

1、本页面内容包含部分的内容是基于公开信息的合理引用;引用内容仅为补充信息,不代表本站立场。

2、若认为本页面引用内容涉及侵权,请及时与本站联系,我们将第一时间处理。

3、其他媒体/个人如需使用本页面原创内容,需注明“来源:[生知库]”并获得授权;使用引用内容的,需自行联系原作者获得许可。

4、投稿及合作请联系:info@biocloudy.com。