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Louisiana State University Health Science Center, Department of Neurosurgery, Shreveport, USA DOI : 10.5137/1019-5149.JTN.5831-12.0 Hemorrhagic complications of acute leukemia are well described and are a common cause of mortality in these patients. However, to our knowledge, fatal intracerebral hemorrhage (ICH) as an initial presentation of acute lymphocytic leukemia (ALL) has only been reported once. We report a case of previously undiagnosed ALL presenting with ICH. Our patient is a 17-year old male who was found unresponsive several hours after complaining of headache. Initial emergency room evaluation found the patient to have anisocoria with a fixed and dilated right pupil and demonstrated evidence of decorticate posturing. Imaging revealed a large right-sided intraparenchymal hemorrhage, intraventricular hemorrhage, midline shift, and uncal herniation. Laboratory evaluation showed marked leukocytosis with blastic predominance and evidence of disseminated intravascular coagulopathy. Emergent surgical intervention was performed. However, despite evacuation of the hematoma, the patient eventually progressed to clinical brain death. Usually, ICH is seen in ALL patients after the diagnosis has been made. We report a unique case of fatal intracranial hemorrhage as the initial presentation of ALL and discuss the possible management dilemmas to treat such entities. ALL should be kept in the broad differential diagnosis of spontaneous ICH, especially in a young patient with evidence of severe coagulopathy. Keywords : Acute lymphocytic leukemia, Disseminated intravascular coagulation, Subarachnoid hemorrhage, Intracerebral hemorrhage