We present the case of a patient with late neuroborreliosis and a spontaneous temporal lobe hemorrhage. Although ischemic stroke and subarachnoid hemorrhage have been reported in association with Lyme disease, intraparenchymal brain hemorrhage has not been previously described in the course of this disease. The patient is a 48-year old male with a progressive spastic paraparesis of months' duration who presented acute headache, confusion, severe left hemiparesis with sensory deficit and homonymous hemianopsia. A cranial computed tomography scan showed an extensive right temporal lobe hemorrhage with subarachnoid invasion. Brain angiographic and angio-magnetic resonance imaging studies excluded hemorrhage-predisposing vascular abnormalities. Cerebrospinal fluid (CSF) studies disclosed mononuclear pleocytosis with elevated protein levels. Both serum and CSF anti-Borrelia titers were significantly increased, and serum Western Blot showed bands to protein 34 (ops B), 57, 59 and 62. The patient was treated with ceftriaxone for 4 weeks, with a favorable outcome. It is suspected that cause of the hemorrhage was parenchymatous Lyme-associated vascular damage and/or microaneurysmatic rupture.