Movement disorders are common manifestations of human immunodeficiency virus (HIV) infection, and may sometimes be the initial manifestations of HIV infection. Although secondary progressive supranuclear palsy (PSP) due to factors such as paraneoplastic, vascular, and hypoxic-ischemic injury have been reported, PSP associated with HIV infection has received little attention. We describe a patient who displayed a progressive parkinsonian phenotype fulfilling the criteria for PSP over a period of a year. The parkinsonism was the initial manifestation of HIV infection and was eliminated by highly active antiretroviral therapy. This case report indicates that one should consider HIV infection as a cause of PSP-like parkinsonism.