D. Robbins et al., Successful treatment of high titer inhibitors in mild hemophilia a with avoidance of factor VIII and immunosuppressive therapy, AM J HEMAT, 68(3), 2001, pp. 184-188
We describe two patients with mild hemophilia A (MHA) who developed high ti
ter inhibitor (HTI) following intensive recombinant factor VIII (rFVIII) co
ncentrate replacement for surgery and trauma. Intranasal desmopressin was i
nstituted shortly following Immunosuppressive therapy (IST) and activated p
rothrombin complex concentrate (APCC) in one case, and following APCC alone
in the second case. Avoidance of factor VIII (FVIII) coupled with intranas
al desmopressin prophylaxis three times a week resulted In undetectable inh
ibitor levels. Both patients have had no further bleeding episodes and have
been maintained on desmopressin prophylaxis prior to activity for the past
2 to 3 years. Recombinant factor VIIa (rFVIIa) was used successfully prior
to a second surgery In one patient without complication. (C) 2001 Wiley-Li
ss, Inc.