Subperiosteal implant denture therapy was initially introduced in 1942 in Sweden and was subsequently used worldwide for the treatment of fully edentulous maxillary or mandibular arches with advanced bone atrophy. Most authors describe decent success rates for mandibular subperiosteal implants in cases with major bone atrophy but follow-up studies for maxillary subperiosteal implants are not available. Here, we report a case of severe maxillary osteolysis secondary to the placement of a subperiosteal in-house implant. Subperiosteal implants are rarely used today but patients still carrying these devices can be challenging to manage when severe complications occur. New technical advances, including the use of surgical planification and additive manufacturing, may lead to a new interest in subperiosteal implants.