HIV infection or complications of HIV-induced immunodeficiency may affect the central nervous system (CNS). However, vascular cerebral pathologies are very rare, in particular intracerebral arteriovenous malformations (AVM). We report the case of an HIV-infected patient who had a cerebral AVM leading to symptoms such as recurring focal seizures. Only after initiation of potent antiretroviral combination therapy, but not antiretroviral monotherapy or bitherapy, could the viral load be suppressed and immunodeficiency resolved. Two years after the start of highly active antiretroviraL therapy (HAART) total occlusion of the AVM could be demonstrated. Taken together, this case report may demonstrate the potent angiogenic activity of HIV for AVM. Also, this case report might show that inhibition of such a cofactor may lead to resolution of an AVM.