Subperiosteal implants were introduced in the last century. Poor clinical results led those implants to be progressively abandoned. Recently, several Authors suggested a revival of subperiosteal implants as an alternative to regenerative procedures. The purpose of this study was to describe the clinical application of custom-made additively manufactured subperiosteal implant for fixed prosthetic rehabilitation of edentulous maxilla. Plaster models of the upper and the lower arch were scanned, as well as the mock-up. Digital Imaging and Communications in Medicine data obtained from cone beam computed tomography were processed through the thresholding procedure. The design of the subperiosteal implant was drawn on the stereolithographic model and scanned. Once the digital project of the subperiosteal implant was completed, it was sent to additive manufacturing. After the surgery, the patient was strictly monitored for up to 2 years. The outcomes were assessed based on the incurrence of biological and mechanical complications, postoperative complications, and implant survival. The patient did not suffer from postoperative complications. Neither biological nor mechanical complications occurred during the follow-up period. At the end of the study, the implant was still in function. Custom-made subperiosteal implants could be considered as an alternative to regenerative procedures for the rehabilitation of severe bone atrophy. Further studies are needed in the future to confirm the positive outcome.