Osteopetrosis (OP) is a rare genetic metabolic bone disorder characterized by osteoclast dysfunction impairing bone resorption. Together with normal osteoblastic activity this dysfunction promotes intense bone sclerosis with reduction of marrow spaces as well as bone turnover. Maxillofacial complications are common after tooth extraction. Osteomyelitis (OM) of the mandible occurs in around 10% of the cases with OP. The purpose of this paper is to report the first case of OP treated with dental implants, and to describe the protocol used to treat OM that developed after failure of one implant. The treatment of OM included long-term use of antibiotics and marginal bone resection with a piezoelectric device. Similar cases to confirm the efficacy of this treatment are required. Schlagwörter: bone resorption, dental implants, osteomyelitis, osteopetrosis