We report the case of a girl with a severe perianal fistulizing Crohn's dis
ease who during intravenous infusion of cyclosporine developed headache, ge
neral seizures and cortical blindness. Head magnetic resonance imaging show
ed spread, cortical-subcortical nonenhancing signal changes. Full neurologi
cal recovery was achieved in 24 hours. Cyclosporine was stopped and a singl
e dose of infliximab was infused with rapid improvement of the fistula that
at 7 months' follow-up is still closed.