We describe a case of hypertrophic osteoarthropathy (HOA), exclusively located in the left lower leg and secondary to vascular prosthesis infection. Four years before, the patient underwent aortofemoral bifurcation grafting because of a ruptured aortic aneurysm. The investigations revealed prosthesis infection by Pseudomona aeruginosa and Bacteroides thetaiotamicron. The onset of HOA in a patient with a vascular prosthesis can help to achieve an early diagnosis of graft infection. The literature on this uncommon association is reviewed.