Y. Aihara et al., RECOMBINANT IFN-GAMMA TREATMENT OF A PATIENT WITH HYPERIMMUNOGLOBULIN-E SYNDROME TRIGGERED AUTOIMMUNE THROMBOCYTOPENIA, Journal of interferon & cytokine research, 18(8), 1998, pp. 561-563
We report a pediatric patient with hyperimmunoglobulin E syndrome (HIE
S) treated with recombinant IFN-gamma (rIFN-gamma) for 2 1/4 years who
developed autoimmune thrombocytopenia and was positive for serum anti
platelet antibody and antinuclear antibody (ANA). She was then treated
with i.v. methylprednisolone pulse therapy followed by oral immunosup
pressive drugs. With this therapy, her platelet count increased and wa
s maintained within the normal range for more than a year. We retrospe
ctively examined her sera stored at -40 degrees C for ANA and found th
at the ANA level was increased from 1:40 to 1:640 with the rIFN-gamma
therapy. Therefore, we believe that, in this case, rIFN-gamma treatmen
t may have played a crucial role in triggering autoimmune thrombocytop
enia. Furthermore, this case demonstrates that caution must be observe
d in administering rIFN-gamma to genetically predisposed patients.