W. Kreuz et al., IMMUNE TOLERANCE THERAPY IN PEDIATRIC HEMOPHILIACS WITH FACTOR-VIII INHIBITORS - 14 YEARS FOLLOW-UP, Haemophilia, 1(1), 1995, pp. 24-32
We report our clinical experience in the immune tolerance (IT) therapy
of 21 paediatric haemophiliacs with FVIII inhibitor: high responders
(16 HR) received initially FVIII twice daily at a dosage of 50-300 U/k
g/day, 11/16 received a concomitant treatment with activated prothromb
in complex concentrate (100-200 U/kg/day). Low responders (five LR) re
ceived 20-100 FVIII U/kg every second or third day. Inhibitor eliminat
ion was achieved in 19/21 patients in a median time of 4 months in HR
and 1.5 months in LR. The outcome and length of time needed to induce
IT was significantly correlated with FVIII exposure between the first
inhibitor detection and onset of IT therapy and to interruption of IT
therapy. For a rapid elimination of FVIII inhibitors it is important t
o start continuous administration of high-dose FVIII (greater than or
equal to 100 FVIII U/kg/day) before repeated exposure to FVIII, in ord
er to prevent rebooster effects, prolongation of elimination time, and
to reduce expense.