Real-world bleeding rates on emicizumab: the value of using nationwide digital treatment diary data in clinical research

Background: People with hemophilia in the Netherlands log bleeds and infusions through a digital treatment diary. With the current innovations in hemophilia treatments, the use of patient-reported bleeding data will become increasingly important. Objective: To assess real-world bleeding rates on emi...

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Main Authors: Martijn R. Brands, Elisabeth M. Taal, Martijn Oude Voshaar, Mariëtte H.E. Driessens, Caroline M.E. van Veen, Marieke J.H.A. Kruip, Paul L. den Exter, Britta A.P. Laros-van Gorkom, Marjet A. Stein-Wit, Kathelijn Fischer, Stephan Meijer, Karina Meijer, Marlène Beijlevelt, Karin Fijnvandraat, Samantha C. Gouw
Format: Article
Language:English
Published: Elsevier 2025-02-01
Series:Research and Practice in Thrombosis and Haemostasis
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Online Access:http://www.sciencedirect.com/science/article/pii/S247503792500041X
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author Martijn R. Brands
Elisabeth M. Taal
Martijn Oude Voshaar
Mariëtte H.E. Driessens
Caroline M.E. van Veen
Marieke J.H.A. Kruip
Paul L. den Exter
Britta A.P. Laros-van Gorkom
Marjet A. Stein-Wit
Kathelijn Fischer
Stephan Meijer
Karina Meijer
Marlène Beijlevelt
Karin Fijnvandraat
Samantha C. Gouw
author_facet Martijn R. Brands
Elisabeth M. Taal
Martijn Oude Voshaar
Mariëtte H.E. Driessens
Caroline M.E. van Veen
Marieke J.H.A. Kruip
Paul L. den Exter
Britta A.P. Laros-van Gorkom
Marjet A. Stein-Wit
Kathelijn Fischer
Stephan Meijer
Karina Meijer
Marlène Beijlevelt
Karin Fijnvandraat
Samantha C. Gouw
author_sort Martijn R. Brands
collection DOAJ
description Background: People with hemophilia in the Netherlands log bleeds and infusions through a digital treatment diary. With the current innovations in hemophilia treatments, the use of patient-reported bleeding data will become increasingly important. Objective: To assess real-world bleeding rates on emicizumab in a nationwide cohort of people with severe hemophilia A, and assess the value of digital treatment diary data. Methods: People with severe hemophilia A of all ages with and without inhibitors using emicizumab who use the digital treatment diary were included. From 2018 to October 2023, data on bleeds treated with clotting factor concentrate were collected from digital treatment diaries and electronic health records. Mean (95% CI) annualized (joint) bleeding rates were calculated using negative-binomial regression analyses. Proportions of people with zero-treated (joint) bleeds were assessed using Kaplan–Meier survival analysis. We calculated the proportion of all bleeds that were recorded in digital treatment diaries. Results: The 232 included persons (median age, 27 years; IQR, 13-51) who used emicizumab for a median of 27 months (IQR, 14-31 months). The mean treated annualized bleeding rate and annualized joint bleeding rate were 1.5 (CI, 1.3-1.8) and 0.8 (CI, 0.6-1.0), respectively. At 24 weeks, 63% had zero-treated bleeds, and 80% had zero-treated joint bleeds. Of treated bleeds, 67% (310/460) were reported in digital treatment diaries. Conclusion: Bleeding rates among Dutch people with severe hemophilia A using emicizumab were comparable to other real-world studies. We formulated recommendations to improve the quality of patient-reported bleeding data, such as establishing guidelines for recording bleeds and improving interoperability.
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spelling doaj-art-cd461b793d794718ab0806affa6ba9ab2025-08-20T03:18:58ZengElsevierResearch and Practice in Thrombosis and Haemostasis2475-03792025-02-019210271710.1016/j.rpth.2025.102717Real-world bleeding rates on emicizumab: the value of using nationwide digital treatment diary data in clinical researchMartijn R. Brands0Elisabeth M. Taal1Martijn Oude Voshaar2Mariëtte H.E. Driessens3Caroline M.E. van Veen4Marieke J.H.A. Kruip5Paul L. den Exter6Britta A.P. Laros-van Gorkom7Marjet A. Stein-Wit8Kathelijn Fischer9Stephan Meijer10Karina Meijer11Marlène Beijlevelt12Karin Fijnvandraat13Samantha C. Gouw14Department of Pediatric Hematology, Amsterdam UMC location University of Amsterdam, Amsterdam, the NetherlandsDepartment of Clinical Epidemiology, Leiden University Medical Center, Leiden, the Netherlands; HemoNED Foundation, Leiden, the NetherlandsDepartment of Public Health, Erasmus University Medical Center Rotterdam, Rotterdam, the NetherlandsNetherlands Hemophilia Patient Society (NVHP), Nijkerk, the NetherlandsHemoNED Foundation, Leiden, the NetherlandsDepartment of Hematology, Erasmus MC, Erasmus University Medical Center Rotterdam, Rotterdam, the NetherlandsDepartment of Medicine, Thrombosis and Hemostasis, Leiden University Medical Center, Leiden, the NetherlandsDepartment of Hematology, Radboud University Medical Center, Nijmegen, the Netherlands; Hemophilia Treatment Center Nijmegen-Eindhoven-Maastricht, Nijmegen, the NetherlandsDepartment of Pediatrics, University Medical Center Groningen, Groningen, the NetherlandsCenter for Benign Haematology, Thrombosis and Haemostasis, Van Creveldkliniek, University Medical Center Utrecht, Utrecht University, Utrecht, the Netherlands; PedNet Haemophilia Research Foundation, Baarn, the NetherlandsNetherlands Hemophilia Patient Society (NVHP), Nijkerk, the NetherlandsDepartment of Hematology, University Medical Center Groningen, University of Groningen, Groningen, the NetherlandsDepartment of Pediatric Hematology, Amsterdam UMC location University of Amsterdam, Amsterdam, the NetherlandsDepartment of Pediatric Hematology, Amsterdam UMC location University of Amsterdam, Amsterdam, the Netherlands; Department of Molecular Cellular Hemostasis, Sanquin Research and Landsteiner Laboratory, Amsterdam, Netherlands, the NetherlandsDepartment of Pediatric Hematology, Amsterdam UMC location University of Amsterdam, Amsterdam, the Netherlands; Department of Clinical Epidemiology, Leiden University Medical Center, Leiden, the Netherlands; Correspondence Samantha C. Gouw, Department of Pediatric Hematology, Amsterdam UMC location University of Amsterdam, Emma Children’s Hospital, Pediatric Hematology, Meibergdreef 9, 1105 AZ Amsterdam, the Netherlands.Background: People with hemophilia in the Netherlands log bleeds and infusions through a digital treatment diary. With the current innovations in hemophilia treatments, the use of patient-reported bleeding data will become increasingly important. Objective: To assess real-world bleeding rates on emicizumab in a nationwide cohort of people with severe hemophilia A, and assess the value of digital treatment diary data. Methods: People with severe hemophilia A of all ages with and without inhibitors using emicizumab who use the digital treatment diary were included. From 2018 to October 2023, data on bleeds treated with clotting factor concentrate were collected from digital treatment diaries and electronic health records. Mean (95% CI) annualized (joint) bleeding rates were calculated using negative-binomial regression analyses. Proportions of people with zero-treated (joint) bleeds were assessed using Kaplan–Meier survival analysis. We calculated the proportion of all bleeds that were recorded in digital treatment diaries. Results: The 232 included persons (median age, 27 years; IQR, 13-51) who used emicizumab for a median of 27 months (IQR, 14-31 months). The mean treated annualized bleeding rate and annualized joint bleeding rate were 1.5 (CI, 1.3-1.8) and 0.8 (CI, 0.6-1.0), respectively. At 24 weeks, 63% had zero-treated bleeds, and 80% had zero-treated joint bleeds. Of treated bleeds, 67% (310/460) were reported in digital treatment diaries. Conclusion: Bleeding rates among Dutch people with severe hemophilia A using emicizumab were comparable to other real-world studies. We formulated recommendations to improve the quality of patient-reported bleeding data, such as establishing guidelines for recording bleeds and improving interoperability.http://www.sciencedirect.com/science/article/pii/S247503792500041Xemicizumabhemophilia Apatient-generated health dataregistriestelemedicine
spellingShingle Martijn R. Brands
Elisabeth M. Taal
Martijn Oude Voshaar
Mariëtte H.E. Driessens
Caroline M.E. van Veen
Marieke J.H.A. Kruip
Paul L. den Exter
Britta A.P. Laros-van Gorkom
Marjet A. Stein-Wit
Kathelijn Fischer
Stephan Meijer
Karina Meijer
Marlène Beijlevelt
Karin Fijnvandraat
Samantha C. Gouw
Real-world bleeding rates on emicizumab: the value of using nationwide digital treatment diary data in clinical research
Research and Practice in Thrombosis and Haemostasis
emicizumab
hemophilia A
patient-generated health data
registries
telemedicine
title Real-world bleeding rates on emicizumab: the value of using nationwide digital treatment diary data in clinical research
title_full Real-world bleeding rates on emicizumab: the value of using nationwide digital treatment diary data in clinical research
title_fullStr Real-world bleeding rates on emicizumab: the value of using nationwide digital treatment diary data in clinical research
title_full_unstemmed Real-world bleeding rates on emicizumab: the value of using nationwide digital treatment diary data in clinical research
title_short Real-world bleeding rates on emicizumab: the value of using nationwide digital treatment diary data in clinical research
title_sort real world bleeding rates on emicizumab the value of using nationwide digital treatment diary data in clinical research
topic emicizumab
hemophilia A
patient-generated health data
registries
telemedicine
url http://www.sciencedirect.com/science/article/pii/S247503792500041X
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