Observational studyJournal of thrombosis and haemostasis : JTH2026
Integrative modeling to improve bleeding risk prediction in adult female hemophilia A carriers.
Observational study in Journal of thrombosis and haemostasis : JTH, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.
What it found
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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.
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Who cites it
2 citing papers in PubMed.
- [Hereditary disorders of hemostasis in obstetrics 2/2-Anesthesiological aspects of secondary hemostasis].Die Anaesthesiologie · 2026Review
- Focus on Women with Hemophilia and Carriers: How to Address All the Existing Gaps in Diagnosis and Care.Journal of clinical medicine · 2026Review
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
6 authors.
Funding
Abstract
backgroundAlthough bleeding phenotypes and factor (F)VIII activity (FVIII:C) in men with hemophilia A correlate highly with F8 mutations, FVIII:C in female HA carriers is more variable due to mosaicism arising from a second wild-type F8 allele and X-chromosome inactivation (XCI). Conventionally, only female HA carriers with low FVIII:C were considered at risk for bleeding. However, hemostatic FVIII:C levels in females remain unestablished. Moreover, HA carriers with normal FVIII:C levels can also have reproductive tract and other bleeding sequelae.
objectivesWe sought to develop a statistical model for predicting abnormal bleeding risk.
methodsPotential HA carriers were enrolled in an observational cross-sectional study. F8 genotyping confirmed the carrier status. Bleeding severity was quantified by bleeding assessment tools. FVIII levels were assessed using 1-stage and chromogenic activity assays, as well as FVIII antigen ELISA. XCI skewing was also quantified. Logistic regression analyses were performed to determine the relationship between abnormal bleeding score and covariates.
resultsOf 92 adult females, 62 were confirmed F8 mutation carriers; 55% of HA carriers had abnormal bleeding (Condensed Molecular and Clinical Markers for the Diagnosis and Management of Type 1 VWD Bleeding Questionnaire 1 score ≥ 4), despite only 19% with FVIII:C < 40%. Logistic regression modeling incorporating XCI skewing, FVIII antigen, and activity performed better than those with FVIII:C alone. Highest model performance was seen for carriers with nonsevere F8 mutations.
conclusionBleeding tendency in HA carriers is common, yet FVIII:C predicts only 38% of those with abnormal bleeding. Logistic regression modeling is highly promising and demonstrates superior performance in predicting bleeding risk compared with baseline FVIII activity alone.
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