A seven year old Hispanic girl developed headache, 
backache and vomiting. A computed tomography showed 2 
intracranial masses. A complete blood count was normal. 
Bone marrow aspiration showed hypoplastic but otherwise 
normal marrow. However, cerebrospinal fluid contained 
leukemic myeloblasts. Four and a half years earlier the 
patient had presented with an orbital mass, lethargy, and 
loss of appetite. MRI demonstrated a mass in left maxillary 
sinus extending to the left orbital cavity causing left 
proptosis. Bone marrow aspiration was diagnostic of Acute 
Myelogenous Leukemia (AML). She was treated with the 
standard AML chemotherapy regimen. Patient achieved 
complete remission and remained in remission during the 
subsequent 4 and a half years until the above event.  
AML recurrence after 4 years of remission is rare. In 
addition this patient initially showed the recurrence only in 
the extramedullary site which is also unusual.  We 
postulate that the intracranial recurrence was due to late 
activation of latent leukemic cells that had seeded in the 
leptomeningeal space at the time of original presentation. 
Because of these 2 unusual aspects we wish to report this 
case.