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The past few years have seen a progressive increment in the number of osteomyelitis cases associated with dental implants, raising the interest of a possible role of implant therapy in the development of osteomyelitis. The aim of the present study was to systematically review the association between dental implant therapy and occurrence of osteomyelitis.The focused question addressed was “What is the risk to develop osteomyelitis among patients receiving dental implants?” Indexed databases were searched without language restrictions up to January 2017 using various key words including: “osteomyelitis”; “dental implants”; “osseointegration”; and “risk factors.”Fourteen studies reporting cases of 39 patients who developed osteomyelitis after dental implant placement were identified. Among the 39 patients, 66.6% were women and 28.2% were men. The overall mean age was 60.26 years. Thirty-six patients had osteomyelitis of the mandible; 2 cases were reported in the maxilla, whereas, 1 case reported vertebral osteomyelitis associated with implant therapy.The knowledge of the real impact of osteomyelitis on the outcome of implant therapy and the identification of risk factors associated with this infectious and life-threatening condition are essential for the development of prevention protocols and treatment strategies.