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Inhibitors to factor VII in congenital factor VII deficiency

FVIII inhibitors are more likely to have uncommon FVIII haplotypes not seen in caucasians. Our patient was hemizygous for FVIII haplotype H2 and his donor was heterozygous for H1 and H2. We speculate that ‘mismatch’ of FVIII haplotypes between donor and recipient might have contributed to inhibitor development, but we cannot prove this in a chimeric immune system that was in part from a donor heterozygous for FVIII H1 and H2 haplotypes. Regardless of the mechanism by which the inhibitor arose in our patient, this case serves as a useful example of successful management of bleeding with OBI-1 and subsequent elimination of the inhibitor without disrupting his stem cell transplant. Acknowledgements