Intraosseous lipoma is an extremely rare variant of lipoma. It is most commonly found in the appendicular skeleton and only very rarely in skull base. We present a 38-year-old male patient who was admitted to our clinic with the symptoms of nasal obstruction and headache. Computed tomography (CT) showed a hyperdense area with surrounding calcification was demonstrated in the anteromedial part of ethmoid sinus. Endoscopic left anterior ethmoidectomy was performed and the mass was totally excised. Histopathological examination was consistent with Milgram's stage 1 intraosseous lipoma. To our knowledge, this is the first reported case of isolated intraosseous lipoma of the ethmoid sinuses in the literature. © 2013 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.