Cerebral dural sinus thrombosis (CSDT) is a rare disease with a high rate of mortality. Until now it has not been well-documented in patients with the acquired immunodeficiency syndrome (AIDS). Prompt diagnosis is essential for initiation of suitable treatment, especially in cases with progressive neurological involvement. The diagnostic methods include computed tomography (CT), magnetic resonance imaging (MRI) and conventional angiography. MRI and magnetic resonance angiography (MRA) have become the methods of choice because of their sensitivity and noninvasive nature. An unusual AIDS-patient, who at the time of CSDT presentation had an associated toxoplasmosis infection is described. To our knowledge, this is the first case of CSDT diagnosed by MRI and MRA in an AIDS patient. In the follow-up, MRI and MRA showed the repermeabilitation of the sinus thrombosis. Cerebral dural sinus thrombosis should be considered as a possible cause of neurological decline in a HIV-patient with central nervous system opportunistic infection.