Colorectal cancer screening in patients with inherited bleeding disorders: high cancer detection rate in hemophilia patients

Eva K. Kempers; Calvin B. van Kwawegen; Joke de Meris; Manon C.W. Spaander; Saskia E.M. Schols; Paula F. Ypma; Floor C.J.I. Heubel-Moenen; Lize F.D. van Vulpen; Michiel Coppens; Johanna G. van der Bom; Karin Fijnvandraat; Karina Meijer; Jeroen Eikenboom; Samantha C. Gouw; Frank W.G. Leebeek; Marieke J.H.A. Kruip

doi:10.1016/j.jtha.2022.12.004

Colorectal cancer screening in patients with inherited bleeding disorders: high cancer detection rate in hemophilia patients

Eva K. Kempers, Calvin B. van Kwawegen, Joke de Meris, Manon C.W. Spaander, Saskia E.M. Schols, Paula F. Ypma, Floor C.J.I. Heubel-Moenen, Lize F.D. van Vulpen, Michiel Coppens, Johanna G. van der Bom, Karin Fijnvandraat, Karina Meijer, Jeroen Eikenboom, Samantha C. Gouw, Frank W.G. Leebeek, Marieke J.H.A. Kruip^*

^*Corresponding author for this work

Research output: Contribution to journal › Article › Academic › peer-review

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Abstract

Background: The population-based colorectal cancer (CRC) screening program in individuals aged 55 to 75 years in the Netherlands uses fecal immunochemical testing (FIT), to detect hemoglobin in feces, followed by colonoscopy in individuals with a positive FIT. Objectives: The objectives of this study are to assess the false-positive rate, detection rate, and positive predictive value of FIT for CRC and advanced adenoma (AA) in patients with Von Willebrand disease (VWD) or hemophilia. Methods: We performed a multicenter, nationwide cross-sectional study embedded in 2 nationwide studies on VWD and hemophilia in the Netherlands. Results: In total, 493 patients with hemophilia (n = 329) or VWD (n = 164) were included, of whom 351 patients participated in the CRC screening program (71.2%). FIT positivity and false-positive rate in patients with hemophilia and VWD were significantly higher than those in the general population (14.8% vs. 4.3%, p <.001 and 10.3% vs. 2.3%, p <.001, respectively). In patients with hemophilia, the detection rate of CRC/AA was significantly higher than that in the general male population (4.5% vs. 1.8%, p =.02), and the positive predictive value of FIT for CRC/AA was comparable (32.3% vs. 39.7%, n.s.). In patients with VWD, the detection rate was similar to that of the general population (0.8% vs. 1.4%, n.s.), whereas the positive predictive value was significantly lower than that in the general population (6.3% vs. 36.8%, p =.02). Conclusion: This study indicates that despite a high false-positive rate of FIT in patients with inherited bleeding disorders, the detection rate of CRC and/or AA in hemophilia patients is high. FIT performs different in patients with hemophilia or VWD compared with the general population.

Original language	English
Pages (from-to)	1177-1188
Number of pages	12
Journal	Journal of Thrombosis and Haemostasis
Volume	21
Issue number	5
Early online date	13 Dec 2022
DOIs	https://doi.org/10.1016/j.jtha.2022.12.004
Publication status	Published - May 2023

Bibliographical note

ACKNOWLEDGMENTS:
The WiN-Pro study was supported by research funding from Stichting Haemophilia (Dutch Haemophilia Foundation), CSL Behring , and Shire . The HiN6 study was supported by research funding from the Dutch Ministry of Health , Welfare , and Sport (Dutch: Ministerie van Volksgezondheid, Welzijn en Sport; VWS) and Stichting Haemophilia (Dutch Haemophilia Foundation). We would like to thank V. Schmidt, E.C. van Balen, and S. Hassan for the collection of data for the HiN6 study. The authors also thank the National Institute for Public Health and the Environment for making data from the national monitoring of the CRC screening program available.

Publisher Copyright: © 2022 The Authors

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Kempers, E. K., van Kwawegen, C. B., de Meris, J., Spaander, M. C. W., Schols, S. E. M., Ypma, P. F., Heubel-Moenen, F. C. J. I., van Vulpen, L. F. D., Coppens, M., van der Bom, J. G., Fijnvandraat, K., Meijer, K., Eikenboom, J., Gouw, S. C., Leebeek, F. W. G., & Kruip, M. J. H. A. (2023). Colorectal cancer screening in patients with inherited bleeding disorders: high cancer detection rate in hemophilia patients. Journal of Thrombosis and Haemostasis, 21(5), 1177-1188. Advance online publication. https://doi.org/10.1016/j.jtha.2022.12.004

@article{720569d5313f4656a2756adc2df43179,

title = "Colorectal cancer screening in patients with inherited bleeding disorders: high cancer detection rate in hemophilia patients",

abstract = "Background: The population-based colorectal cancer (CRC) screening program in individuals aged 55 to 75 years in the Netherlands uses fecal immunochemical testing (FIT), to detect hemoglobin in feces, followed by colonoscopy in individuals with a positive FIT. Objectives: The objectives of this study are to assess the false-positive rate, detection rate, and positive predictive value of FIT for CRC and advanced adenoma (AA) in patients with Von Willebrand disease (VWD) or hemophilia. Methods: We performed a multicenter, nationwide cross-sectional study embedded in 2 nationwide studies on VWD and hemophilia in the Netherlands. Results: In total, 493 patients with hemophilia (n = 329) or VWD (n = 164) were included, of whom 351 patients participated in the CRC screening program (71.2%). FIT positivity and false-positive rate in patients with hemophilia and VWD were significantly higher than those in the general population (14.8% vs. 4.3%, p <.001 and 10.3% vs. 2.3%, p <.001, respectively). In patients with hemophilia, the detection rate of CRC/AA was significantly higher than that in the general male population (4.5% vs. 1.8%, p =.02), and the positive predictive value of FIT for CRC/AA was comparable (32.3% vs. 39.7%, n.s.). In patients with VWD, the detection rate was similar to that of the general population (0.8% vs. 1.4%, n.s.), whereas the positive predictive value was significantly lower than that in the general population (6.3% vs. 36.8%, p =.02). Conclusion: This study indicates that despite a high false-positive rate of FIT in patients with inherited bleeding disorders, the detection rate of CRC and/or AA in hemophilia patients is high. FIT performs different in patients with hemophilia or VWD compared with the general population.",

author = "Kempers, {Eva K.} and {van Kwawegen}, {Calvin B.} and {de Meris}, Joke and Spaander, {Manon C.W.} and Schols, {Saskia E.M.} and Ypma, {Paula F.} and Heubel-Moenen, {Floor C.J.I.} and {van Vulpen}, {Lize F.D.} and Michiel Coppens and {van der Bom}, {Johanna G.} and Karin Fijnvandraat and Karina Meijer and Jeroen Eikenboom and Gouw, {Samantha C.} and Leebeek, {Frank W.G.} and Kruip, {Marieke J.H.A.}",

note = "ACKNOWLEDGMENTS: The WiN-Pro study was supported by research funding from Stichting Haemophilia (Dutch Haemophilia Foundation), CSL Behring , and Shire . The HiN6 study was supported by research funding from the Dutch Ministry of Health , Welfare , and Sport (Dutch: Ministerie van Volksgezondheid, Welzijn en Sport; VWS) and Stichting Haemophilia (Dutch Haemophilia Foundation). We would like to thank V. Schmidt, E.C. van Balen, and S. Hassan for the collection of data for the HiN6 study. The authors also thank the National Institute for Public Health and the Environment for making data from the national monitoring of the CRC screening program available. Publisher Copyright: {\textcopyright} 2022 The Authors",

year = "2023",

month = may,

doi = "10.1016/j.jtha.2022.12.004",

language = "English",

volume = "21",

pages = "1177--1188",

journal = "Journal of Thrombosis and Haemostasis",

issn = "1538-7933",

publisher = "Wiley-Blackwell Publishing Ltd",

number = "5",

}

Kempers, EK , van Kwawegen, CB, de Meris, J, Spaander, MCW, Schols, SEM, Ypma, PF, Heubel-Moenen, FCJI, van Vulpen, LFD, Coppens, M, van der Bom, JG, Fijnvandraat, K, Meijer, K, Eikenboom, J, Gouw, SC, Leebeek, FWG & Kruip, MJHA 2023, 'Colorectal cancer screening in patients with inherited bleeding disorders: high cancer detection rate in hemophilia patients', Journal of Thrombosis and Haemostasis, vol. 21, no. 5, pp. 1177-1188. https://doi.org/10.1016/j.jtha.2022.12.004

TY - JOUR

T1 - Colorectal cancer screening in patients with inherited bleeding disorders

T2 - high cancer detection rate in hemophilia patients

AU - Kempers, Eva K.

AU - van Kwawegen, Calvin B.

AU - de Meris, Joke

AU - Spaander, Manon C.W.

AU - Schols, Saskia E.M.

AU - Ypma, Paula F.

AU - Heubel-Moenen, Floor C.J.I.

AU - van Vulpen, Lize F.D.

AU - Coppens, Michiel

AU - van der Bom, Johanna G.

AU - Fijnvandraat, Karin

AU - Meijer, Karina

AU - Eikenboom, Jeroen

AU - Gouw, Samantha C.

AU - Leebeek, Frank W.G.

AU - Kruip, Marieke J.H.A.

N1 - ACKNOWLEDGMENTS: The WiN-Pro study was supported by research funding from Stichting Haemophilia (Dutch Haemophilia Foundation), CSL Behring , and Shire . The HiN6 study was supported by research funding from the Dutch Ministry of Health , Welfare , and Sport (Dutch: Ministerie van Volksgezondheid, Welzijn en Sport; VWS) and Stichting Haemophilia (Dutch Haemophilia Foundation). We would like to thank V. Schmidt, E.C. van Balen, and S. Hassan for the collection of data for the HiN6 study. The authors also thank the National Institute for Public Health and the Environment for making data from the national monitoring of the CRC screening program available. Publisher Copyright: © 2022 The Authors

PY - 2023/5

Y1 - 2023/5

N2 - Background: The population-based colorectal cancer (CRC) screening program in individuals aged 55 to 75 years in the Netherlands uses fecal immunochemical testing (FIT), to detect hemoglobin in feces, followed by colonoscopy in individuals with a positive FIT. Objectives: The objectives of this study are to assess the false-positive rate, detection rate, and positive predictive value of FIT for CRC and advanced adenoma (AA) in patients with Von Willebrand disease (VWD) or hemophilia. Methods: We performed a multicenter, nationwide cross-sectional study embedded in 2 nationwide studies on VWD and hemophilia in the Netherlands. Results: In total, 493 patients with hemophilia (n = 329) or VWD (n = 164) were included, of whom 351 patients participated in the CRC screening program (71.2%). FIT positivity and false-positive rate in patients with hemophilia and VWD were significantly higher than those in the general population (14.8% vs. 4.3%, p <.001 and 10.3% vs. 2.3%, p <.001, respectively). In patients with hemophilia, the detection rate of CRC/AA was significantly higher than that in the general male population (4.5% vs. 1.8%, p =.02), and the positive predictive value of FIT for CRC/AA was comparable (32.3% vs. 39.7%, n.s.). In patients with VWD, the detection rate was similar to that of the general population (0.8% vs. 1.4%, n.s.), whereas the positive predictive value was significantly lower than that in the general population (6.3% vs. 36.8%, p =.02). Conclusion: This study indicates that despite a high false-positive rate of FIT in patients with inherited bleeding disorders, the detection rate of CRC and/or AA in hemophilia patients is high. FIT performs different in patients with hemophilia or VWD compared with the general population.

AB - Background: The population-based colorectal cancer (CRC) screening program in individuals aged 55 to 75 years in the Netherlands uses fecal immunochemical testing (FIT), to detect hemoglobin in feces, followed by colonoscopy in individuals with a positive FIT. Objectives: The objectives of this study are to assess the false-positive rate, detection rate, and positive predictive value of FIT for CRC and advanced adenoma (AA) in patients with Von Willebrand disease (VWD) or hemophilia. Methods: We performed a multicenter, nationwide cross-sectional study embedded in 2 nationwide studies on VWD and hemophilia in the Netherlands. Results: In total, 493 patients with hemophilia (n = 329) or VWD (n = 164) were included, of whom 351 patients participated in the CRC screening program (71.2%). FIT positivity and false-positive rate in patients with hemophilia and VWD were significantly higher than those in the general population (14.8% vs. 4.3%, p <.001 and 10.3% vs. 2.3%, p <.001, respectively). In patients with hemophilia, the detection rate of CRC/AA was significantly higher than that in the general male population (4.5% vs. 1.8%, p =.02), and the positive predictive value of FIT for CRC/AA was comparable (32.3% vs. 39.7%, n.s.). In patients with VWD, the detection rate was similar to that of the general population (0.8% vs. 1.4%, n.s.), whereas the positive predictive value was significantly lower than that in the general population (6.3% vs. 36.8%, p =.02). Conclusion: This study indicates that despite a high false-positive rate of FIT in patients with inherited bleeding disorders, the detection rate of CRC and/or AA in hemophilia patients is high. FIT performs different in patients with hemophilia or VWD compared with the general population.

UR - http://www.scopus.com/inward/record.url?scp=85150260582&partnerID=8YFLogxK

U2 - 10.1016/j.jtha.2022.12.004

DO - 10.1016/j.jtha.2022.12.004

M3 - Article

C2 - 36696188

AN - SCOPUS:85150260582

SN - 1538-7933

VL - 21

SP - 1177

EP - 1188

JO - Journal of Thrombosis and Haemostasis

JF - Journal of Thrombosis and Haemostasis

IS - 5

ER -

Colorectal cancer screening in patients with inherited bleeding disorders: high cancer detection rate in hemophilia patients

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