Bacillary angiomatosis, a recently described illness that occurs predominantly in patients infected with human immunodeficiency virus (HIV), has a wide spectrum of manifestations. Intracerebral lesions, however, have not yet been reported. We describe a 49-year-old HIV-positive man with cutaneous and intracerebral bacillary angiomatosis. The patient initially presented with an intracranial mass lesion and, after an extensive evaluation that included two brain biopsies, the cause remained unknown. We subsequently diagnosed cutaneous bacillary angiomatosis, which led to a retrospective diagnosis of intracerebral bacillary angiomatosis. The skin and brain lesions had typical features of bacillary angiomatosis; histologic vasoproliferative inflammatory responses and clumps of pleomorphic bacilli observed with Warthin-Starry staining. In addition, both lesions improved dramatically with erythromycin therapy. This case expands the spectrum of bacillary angiomatosis and identifies a new case of treatable brain lesions in HIV-inspected patients.