Acquired haemophilia A: A 15-year population-based review of incidence rate, patient demographics and treatment outcomes

Haemophilia. 2023 Sep;29(5):1269-1275. doi: 10.1111/hae.14845. Epub 2023 Aug 28.

Abstract

Introduction: Acquired haemophilia A (AHA) is a rare bleeding disorder characterized by autoantibodies against coagulation factor VIII (FVIII). Estimates of AHA incidence are largely based on registry data, which may be prone to referral bias. Population-based studies can enhance our understanding of the epidemiology, presentation and outcomes of AHA.

Methods: We conducted a retrospective, population-based cohort study of all AHA diagnosed and treated in Manitoba, Canada over a 15-year period. Using records from the sole provincial reference laboratory, we identified all patients with FVIII inhibitors who did not have congenital haemophilia. Using a piloted case report form, patient data was ascertained from hospital and bleeding disorder clinic records.

Results: From 2006 to 2021, we identified 34 patients with AHA, corresponding to a population-based incidence rate of AHA of 1.78 cases per million per year. The median age at presentation was 76 years and most cases were idiopathic (79%). Almost all patients (97%) presented with bleeding, of which 58% were considered major bleeds and required haemostatic agents in 67%. Longstanding unexplained bleeding symptoms were commonly reported, suggesting delayed diagnosis. Immunosuppressive therapy (IST) was administered in 88% of patients. Remission was achieved in 79% of patients; median time to remission was 2.1 months. There were two deaths due to bleeding. No deaths due to IST were reported.

Conclusion: The population-based incidence of AHA in Manitoba is 1.78 cases/million/year. Bleeding is common and can be life-threatening. AHA outcomes are encouraging with the use of haemostatic agents and IST. Serious treatment-associated morbidity and mortality is uncommon.

Keywords: acquired haemophilia; bleeding disorders; cohort study; epidemiology; population-based.

MeSH terms

  • Cohort Studies
  • Hemophilia A* / drug therapy
  • Hemophilia A* / epidemiology
  • Humans
  • Incidence
  • Retrospective Studies
  • Treatment Outcome

Supplementary concepts

  • Factor 8 deficiency, acquired