Priapism is defined as a prolonged penile erection of more than 6 h in the absence of sexual stimulation, which persists despite orgasm. The classification into two broad subtypes, low-flow and high-flow priapism, is universal. Stuttering priapism refers to a distinct condition whereby patients develop short-lived self-limiting episodes of prolonged and painful erections [1,2]. We report a rare case of stuttering priapism in a HIV positive patient. A 60-year-old white male with a long-standing HIV/HBV coinfection was admitted with an orthostatic stuttering painful penile erection. His current illness started 2 days earlier, when he developed a painful swelling of the lower limbs. There was no history of trauma, recent sexual activity, bicycle riding, alcohol consumption, and no previous priapism. His treatment included lamivudine, ritonavir, darunavir, etravirine, enfuvirtide, and tenofovir. Past medical history revealed left calf deep vein thrombosis and pulmonary embolism related to protein S deficiency 14 years earlier. Treatment at that time was cava filter implantation because of cerebral cavernoma hemorrhage under anticoagulation therapy. Physical examination in supine position was unremarkable with the penis at rest. When the patient stood up, examination showed a man distressed with pain, exhibiting swollen warm, tender lower limbs with tense shiny skin. The penis was markedly engorged and extremely tender. Angiographic computed tomography confirmed the diagnosis of extensive deep vein thrombosis extending into the deep pelvic veins and cava vein with cava filter thrombosis. Other investigations including full blood count, kidney, and liver function tests were all normal. The patient received analgesia and unfractionated heparin intravenously followed by a subcutaneous low-molecular weight heparin (enoxaparin sodium) 80 mg twice daily. On the sixth day of hospitalization, there was a 2 cm decrease in the circumference of both lower limbs, and the penis remained at rest in the supine position. After 26 days in hospital, the patient was discharged in a stable condition. He was maintained on warfarin 12.5 mg/day, achieving an international normalized ratio (INR) of 3. The majority of our understanding of stuttering priapism is derived from observations in patients with sickle cell disease. These episodes are commonly short lived (<3 h) and are self-limiting, although some are prolonged and require acute medical intervention [2]. Physicians should be aware of this possible complication.