Both akinetic and hyperkinetic movement disorders may rarely be the presenting feature of human immunodeficiency virus (HIV) infection. The possible pathogenic basis is the involvement of subcortical structures by the HIV infection-related pathology. Opportunistic infections, or mass lesions complicating HIV infection. In addition dopaminergic dysfunction and medications may also play a role. We report a HIV infected male who presented with progressive choreoathetoid movements and dystonia. He had remarkable improvement of the movement disorder with tetrabenazine and anti-retroviral therapy (HAART) treatment.