Report of a patient with chronic intractable autoimmune hemorrhaphilia due to anti-factor XIII/13 antibodies who died of hemorrhage after sustained clinical remission for 3 years

Int J Hematol. 2015 Jun;101(6):598-602. doi: 10.1007/s12185-015-1754-8. Epub 2015 Feb 8.

Abstract

Although the incidence of autoimmune hemorrhaphilia due to anti-Factor XIII (FXIII, not FVIII or FXII to avoid confusion) antibodies (AH13) or hemorrhagic "acquired FXIII deficiency due to anti-FXIII autoantibodies" was previously considered rare, it has been on the increase in the twenty-first century, at least in Japan. An 83-year-old woman with an unexplained hemorrhage was admitted to our hospital for intramuscular hematoma and severe anemia. Her FXIII activity was reduced to 10 % of normal; since FXIII inhibitors and anti-FXIII-A subunit autoantibodies were detected, she was definitively diagnosed with AH13. Despite developing cardiac tamponade due to pericardial hemorrhage, she clinically recovered from AH13 after hemostatic therapy with FXIII-concentrates and immunosuppressive treatment with rituximab and cyclophosphamide. However, her FXIII activity remained low and she died of hemorrhage 3.5 years after admission. AH13 patients should be monitored for a prolonged period, as this disease is very likely a chronic intractable hemorrhagic disorder.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Aged, 80 and over
  • Autoantibodies / immunology*
  • Autoimmune Diseases / complications
  • Autoimmune Diseases / drug therapy*
  • Autoimmune Diseases / immunology
  • Chronic Disease
  • Cyclophosphamide / therapeutic use
  • Factor XIII / immunology*
  • Factor XIII Deficiency / complications
  • Factor XIII Deficiency / drug therapy*
  • Factor XIII Deficiency / immunology
  • Female
  • Hematoma / complications
  • Hematoma / drug therapy
  • Hematoma / immunology
  • Hemorrhage / complications
  • Hemorrhage / drug therapy*
  • Hemorrhage / immunology
  • Hemostatics / therapeutic use*
  • Humans
  • Immunosuppressive Agents / therapeutic use*
  • Remission Induction
  • Rituximab / therapeutic use

Substances

  • Autoantibodies
  • Hemostatics
  • Immunosuppressive Agents
  • Rituximab
  • Cyclophosphamide
  • Factor XIII