Successful induction of immune tolerance in a patient with haemophilia B with inhibitor.
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.
Suzuki
N
N
Department of Pediatrics, Sapporo Medical University School of Medicine, Chuo-Ku, Sapporo, Japan. nsuzuki@sapmed.ac.jp
Watanabe
J
J
Kudoh
T
T
Hori
T
T
Hatakeyama
N
N
Mizue
N
N
Oda
T
T
Tsutsumi
H
H
D004334:Drug Administration Schedule; D003875:Drug Eruptions / Q000209:etiology; D005164:Factor IX / Q000009:adverse effects / Q000037:antagonists & inhibitors* / Q000627:therapeutic use; D002836:Hemophilia B / Q000188:drug therapy* / Q000276:immunology; D006801:Humans; D007108:Immune Tolerance; D007223:Infant; D008297:Male; D014581:Urticaria / Q000139:chemically induced
D005164:Factor IX