Evidence map›Paper›PMID 40421900›Full record

ArticleHaemophilia : the official journal of the World Federation of Hemophilia2025

Current Practice Regarding Bleeding Disorders of Unknown Cause in the Netherlands: A National Survey.

Caroline M A Mussert, Amaury L L Monard, Tirsa T van Duijl, Yvonne M C Henskens, Maartje van den Biggelaar, Roger E G Schutgens, Saskia E M Schols, Karin J Fijnvandraat, Karina Meijer, Paul L den Exter and 6 more

Registry-linked trialAbstract read
In one paragraph

Article in Haemophilia : the official journal of the World Federation of Hemophilia, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT07454161 (Bleeding Disorder of Unknown Cause In the Netherlands), which is not on this map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

NCT07454161 recruitingnot on this mapstarted 2026, after this paper: background citation

Bleeding Disorder of Unknown Cause In the Netherlands (BDUC-iN Study)

Typeobservational_patient_registrySponsorMaastricht University Medical CenterRan2026 to 2039Enrolled500ConditionsHemorrhagic Disorders, Bleeding Disorder of Unknown Cause
3 · Its place in the literature

Who cites it

2 citing papers in PubMed.

  1. Seventh Åland Island Meeting on von Willebrand Disease.Haemophilia : the official journal of the World Federation of Hemophilia · 2026
    Article
  2. Observational
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

16 authors.

Caroline M A MussertDepartment of Pediatric Hematology and Oncology, Erasmus MC Sophia Children's Hospital, University Medical Center Rotterdam, Rotterdam, the Netherlands.ORCID https://orcid.org/0009-0009-8970-9473
Amaury L L MonardDepartment of Hematology, Maastricht University Medical Center, Maastricht, the Netherlands.ORCID https://orcid.org/0009-0007-0208-5764
Tirsa T van DuijlDepartment of Molecular Hematology, Sanquin Research, Amsterdam, the Netherlands.
Yvonne M C HenskensCARIM - School for Cardiovascular Disease, Maastricht University, Maastricht, the Netherlands.
Maartje van den BiggelaarDepartment of Molecular Hematology, Sanquin Research, Amsterdam, the Netherlands.
Roger E G SchutgensCenter for Benign Haematology, Thrombosis and Haemostasis, Van Creveldkliniek, University Medical Center Utrecht, Utrecht, the Netherlands.ORCID https://orcid.org/0000-0002-2762-6033
Saskia E M ScholsDepartment Hematology, Radboud University Medical Center, the Netherlands and Hemophilia Treatment Centre Nijmegen-Eindhoven-Maastricht, Nijmegen, the Netherlands.
Karin J FijnvandraatDepartment Pediatric Hematology, Amsterdam UMC Emma Children's Hospital, University of Amsterdam, Amsterdam, the Netherlands.
Karina MeijerDepartment Hematology, University Medical Center Groningen, Groningen, the Netherlands.
Paul L den ExterDepartment Vascular Medicine, Leiden University Medical Center, Leiden, the Netherlands.
Laurens NieuwenhuizenDepartment Internal Medicine, Maxima Medical Center, Veldhoven, the Netherlands.
Iris van MoortDepartment Hematology, Erasmus MC, University Medical Center Rotterdam, Rotterdam, the Netherlands.ORCID https://orcid.org/0000-0002-0248-4710
Marieke J H A KruipDepartment Hematology, Erasmus MC, University Medical Center Rotterdam, Rotterdam, the Netherlands.ORCID https://orcid.org/0000-0002-0265-4871
Marjon H CnossenDepartment of Pediatric Hematology and Oncology, Erasmus MC Sophia Children's Hospital, University Medical Center Rotterdam, Rotterdam, the Netherlands.
Floor C J I Heubel-MoenenDepartment of Hematology, Maastricht University Medical Center, Maastricht, the Netherlands.ORCID https://orcid.org/0000-0002-3281-926X
BDUC‐iN study group

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

introductionAbout 40%-70% of persons with a clinically relevant bleeding tendency who are referred to haemostasis experts are classified as having a 'bleeding disorder of unknown cause' (BDUC) as no biological entity can be found after extensive laboratory testing. Currently, guidelines are under development regarding diagnostic assessment and management to minimize variation in clinical practice.

aimInvestigate current practices regarding BDUC in the Netherlands.

methodsAn online survey on the best BDUC definition, associated bleeding phenotype, clinical and diagnostic approaches, treatment, registration, and follow-up was distributed amongst healthcare providers working in Dutch haemophilia treatment centres (HTCs).

resultsThe survey was completed by 39/54 (72%) respondents. Twenty percent did not register BDUC patients in their HTC. Healthcare professionals indicated that follow-up should depend on bleeding phenotype severity and bleeding history, and other potential causes for an increased bleeding tendency should be excluded. Moreover, the use of laboratory tests within the routine diagnostic pathway was demonstrated to be heterogeneous. Regarding treatment, tranexamic acid was most frequently prescribed for minor and major surgical interventions (79% and 86%), dental extractions (93%) and childbirth (93%). Desmopressin was prescribed for major surgical procedures by 79%.

conclusionOur survey shows that Dutch current practice varies but is generally in line with recent ISTH SSC recommendations. Additionally, it describes other clinically relevant topics not included in the international survey, such as follow-up and exclusion of other causes for bleeding. This survey therefore adds to international efforts to unify BDUC definition, diagnostic approach, treatment and follow-up, and to attain broadly supported guidelines.

Indexed as

Blood Coagulation DisordersHemorrhagic DisordersHumansNetherlandsSurveys and QuestionnairesTranexamic AcidTranexamic Acidbleeding disorder of unknown causecurrent practicediagnosticshaemostasismanagement

Identifiers

PMID40421900
PMCPMC12311882

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Registered trials

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.