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  • Title: [Successful induction of immune tolerance and novel hemostatic effects in a hemophilia A with high-responder inhibitor by regular infusions of factor VIII].
    Author: Shima M, Sawamoto Y, Kamisue M, Shibata Y, Tuzi S, Kuwabara M, Tanaka I, Tanizawa T, Tanaka A, Ueda M, Kakishita E, Yoshioka A.
    Journal: Rinsho Ketsueki; 1996 Nov; 37(11):1303-8. PubMed ID: 8960666.
    Abstract:
    A hemophilia A patient with high responder inhibitor had been treated by (activated) prothrombin complex concentrates (A) PCC and activated factor VII until the occurrence of intracranial bleeding at the age of 6 years. Since the inhibitor titer was decreased less than 1 Bethesda Units/ml, high dose of factor VIII was given followed by the infusions of factor VIII concentrates (100 units/kg) three times a week. In spite of previous episodes of anamnestic responses by factor VIII products before, the inhibitor titer did not increase and disappeared completely 6 months after the FVIII infusion therapy. The specific anti-factor VIII IgG subclasses of the inhibitor were IgG2 and IgG4. The inhibitor recognized both light and heavy chains. He have no bleeding episode for 6 months since the beginning of the prophylactic with factor VIII concentrates.
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