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An infected vascular graft is an uncommon association in hypertrophic osteoarthropathy (HOA) but is important because of potential catastrophic complications. Here we describe 3 new patients with HOA secondary to vascular prosthesis infection and review the other 23 patients described in the English literature with this clinical syndrome. The finding of symptoms and signs of localized HOA in patients with arterial prosthesis may be the presenting manifestation of graft sepsis. In most patients the graft is located at the aorta or aortic bifurcation. The initial symptoms are fever, joint pain mainly involving the knees and ankles, clubbing, and in some cases intestinal bleeding. Recurrent abscesses and bacteremia arising in spite of appropriate antimicrobial therapy are prominent infectious signs. Another characteristic feature is the wide variety of bacteria isolated from a single patient. When a polymicrobial enteric flora is isolated from the blood or other tissue, an intestinal origin of infection can be readily suspected. Gallium or leukocyte scans are useful for detecting an abscess around the vascular prosthesis. In some cases, the detection of gas around the graft demonstrated by computed tomography is highly suggestive of prosthesis infection. Mechanisms involved in the pathogenesis of HOA associated with aortic graft infection are unknown. Treatment consists of intravenous antibiotics and surgical replacement of the infected graft. The prognosis of arterial prosthesis infection depends greatly on early diagnosis and treatment before the onset of life-threatening complications. Knowledge of this rare association may lead to an early diagnosis and appropriate management of this serious and often fatal complication.