We report the case of an 18 year-old woman treated for femoral osteoge
nic sarcoma who presented generalized seizures requiring sedation, tra
cheal intubation and artificial ventilation. CT brain scan showed diff
use hypodensities. Doppler studies showed an increased cerebral arteri
al resistance. Regional cerebral blood flow was decreased. A right car
otid angiogram showed abnormalities consistent with diffuse cerebral a
rteritis. The patient slowly recovered and 6 weeks later, magnetic res
onance imaging showed disseminated areas of hyposignal on T1 and hyper
signal on T2 weighted images. We reviewed the different published case
s of acute high dose methotrexate neurotoxicity and the different unde
rlying mechanisms.