Background: Maxillary sinus augmentation is a method extensively used to restore sufficient bone volume in the posterior maxilla to allow for the placement of fixtures. The purpose of the present case report was to describe a rare case of sinus osteoma used for implant support and to review the relevant literature. Materials and Methods: A 58-year-old man with a radiopaque intrasinusal lesion was referred for rehabilitation of the maxilla. The lesion was probably an osteoma and involved the nasal wall of the maxillary sinus. After discussing the options with the patient, he agreed to maintain the lesion and a sinus augmentation with a bone graft. A part of the osteoma was partially removed for histological analysis while avoiding perforation or tearing of the schneiderian membrane. After six months, 6 implants (Bone System Implant, Milano, Italy) were placed in the maxilla, two of which were inserted in the osteoma. Results: The two implants placed in the osteoma were perfectly osseointegrated. The graft material appeared well-integrated with no local signs of inflammation. No postoperative events or symptoms were reported after the surgery stages and at a 6-month follow-up. Regarding the two implants placed in the osteoma: article selection identified 9 case reports, 2 case series, and 1 retrospective study for a total of 58 subjects, 35 males and 25 females. The patients’ ages were heterogeneous and ranged between 12 and 79 years old. Conclusions: In the present case, we decided to leave the osteoma because it was asymptomatic and used as dental implant support. The effectiveness of the present investigation can provide useful guidance for surgeons and dentists in the management of similar clinical situations.