Intravascular papillary endothelial hyperplasia (IPEH) is an unusual, benign, non-neoplastic, vascular lesion characterized histologically by papillary fronds lined by proliferating endothelium. Osteoma is a benign osteogenic lesion characterized by proliferation of compact or cancellous bone. Osteoma of the maxillary sinus is an uncommon lesion. The purpose of this report is to present a case with 2 different lesions (intravascular papillary endothelial hyperplasia and osteoma) occurring at the same time in 1 patient that may or may not be related to each other and to stress the value of diagnostic imaging.