Intravascular papillary endothelial hyperplasia (IVPEH) is an unusual form of intravascular endothelial proliferation. Fine needle aspiration (FNA) diagnosis of IVPEH is quite challenging and only rare reports of the cytopathological features of this entity have been published. We report a case of a 55-year-old female patient who presented with a mass on her left jaw. FNA of the mass revealed pleomorphic polygonal and spindle cells. A preliminary (onsite) cytological diagnosis of suspicious for malignancy was rendered. Subsequent cell block showed delicate papillae composed of attenuated endothelial cells overlying collagenized cores. The endothelial cells were positive for CD34 and factor VIII, supporting the cytological diagnosis of IVPEH. Surgical excision confirmed the diagnosis. To the best of our knowledge, this is the first reported case of IVPEH diagnosed by preoperative FNA.