Background The aim of the present report was to present the clinical, radiological and histological outcomes of a case of implant placement in the upper jaw of a patient affected by osteopetrosis. Case report In a 68 years old patient, 2 implants were placed in a portion of bone with areas with high density bone. Histological evaluation confirmed the radiological diagnosis of osteopetrosis. Implants were loaded 8 months after their placement. No complications or adverse reactions were observed after a 24-month follow up. Conclusion Within the limitations of the present report, it can be concluded that a successful implant rehabilitation can be done in patients affected by osteopetrosis. Further studies, on a larger sample size, are needed to confirm this result