Ectopic pituitary neuroendocrine tumors (PitNETs) are uncommon conditions that develop outside of the sella turcica. The sphenoid sinus is the most common site for ectopic PitNET, followed by the suprasellar region, clivus, and cavernous sinus. PitNETs, regardless of whether inside or outside sella, may display avid 18F-fluorodeoxyglucose (FDG) uptake and masquerade as malignant tumors. Herein, we report a case of ectopic PitNET arising in the sphenoid sinus, which was found as an FDG-avid mass during cancer screening. On magnetic resonance imaging, the tumor showed heterogeneous and intermediate signal intensity areas on T1- and T2-weighted images with cystic components, which was consistent with PitNET. The localization and the presence of empty sella were suggestive of ectopic PitNET, and the diagnosis of ectopic PitNET (prolactinoma) was confirmed by endoscopic biopsy. Ectopic PitNET should be considered in a mass similar in nature to an orthogonal PitNET in areas near the sella turcica especially in patients with empty sella.