A case of progressive multifocal leukoencephalopathy associated with human immunodeficiency virus infection with a fatal outcome is presented. The disease has not been reported from our region before. The patient presented initially with hemiparesis and non-enhancing lesion on computed tomography scan that was thought to be an infarct. After a delay of 4 months, the diagnosis was made by brain biopsy. This delay probably contributed to his unfortunate outcome. The case is described, and methods of diagnosis and treatment are discussed.