In immunosuppressed individuals, such as hematopoietic stem cell transplant recipients, adenoviruses (ADVs) are a well-known cause of morbidity and mortality, with limited treatment options. However, only a few cases were reported in patients with acquired immunodeficiency syndrome (AIDS), and little is known about the relevance of such an infection in these patients with many other concomitant opportunistic infections. We report the case of a 34-year-old man with AIDS presenting with gross hematuria, right flank pain, and acute decrease in kidney function superimposed on chronic kidney disease. His CD4 count was 0/muL despite highly active antiretroviral therapy. A computed tomographic scan showed enlargement of the right renal pelvis. Cystoscopy showed no clots or macroscopic lesions. Urine analysis showed no bacteria or abnormal epithelial cells. ADV was found in viral culture and by using real-time polymerase chain reaction in the patient's urine and later in blood. The renal biopsy specimen showed ADV-related tubulointerstitial nephritis with intranuclear inclusions in tubular cells stained by anti-ADV antibodies, in addition to chronic tubular and vascular changes. The ADV serotype belonged to subgroup B. Cidofovir therapy was contraindicated for this patient; therefore, he was administered intravenous ribavirin. The efficiency of this treatment could not be assessed because he rapidly developed neutropenia and disseminated aspergillosis and died. This case illustrates another cause of acute kidney disease in very immunosuppressed patients with AIDS, probably underdiagnosed.