Successful induction of immune tolerance in a patient with haemophilia B with inhibitor

Haemophilia. 2003 May;9(3):340-2. doi: 10.1046/j.1365-2516.2003.00754.x.

Abstract

We describe successful induction of immune tolerance (IT) in a 10-month-old boy with severe haemophilia B. Urticaria developed soon after starting prophylactic treatment and was associated with an inhibitor at 7 Bethesda units mL(-1). Initially, we tried low dose factor IX therapy to induce IT with only a transient effect. The patient experienced an intracranial haemorrhage. A simple bolus dose of FIX eradicated the inhibitor. Thereafter he has been free from inhibitor and nephrotic syndrome for more than 5 years, although he receives FIX three times a week.

Publication types

  • Case Reports

MeSH terms

  • Drug Administration Schedule
  • Drug Eruptions / etiology
  • Factor IX / adverse effects
  • Factor IX / antagonists & inhibitors*
  • Factor IX / therapeutic use
  • Hemophilia B / drug therapy*
  • Hemophilia B / immunology
  • Humans
  • Immune Tolerance*
  • Infant
  • Male
  • Urticaria / chemically induced

Substances

  • Factor IX