A 31-year-old man presented with a 6-month history of a progressive enlargement over the medial right supraorbital margin with intermittent dull pain. His ophthalmologist had diagnosed a sebaceous cyst while the lesion had gradually increased in size. Physical examination revealed a firm, movable mass covered by erythematous skin (Fig. 1A). MRI revealed a 2.5-cm well-circumscribed medial right supraorbital mass showing contrast enhancement with a hyperintense central portion on T2-weighted images and hypointense on T1-weighted images (Fig. 1B,C). Through a skin incision hidden in the eyebrow, the tumor was easily removed sparing the eyelid (Fig. 2A). Histologic examination with hematoxylin and eosin stain showed a solid, well-circumscribed tumor, consisting of bundles of spindle-shaped cells. The immunohistochemical study revealed intense positivity for muscle-specific actin and smooth muscle actin. With all these findings, a diagnosis of leiomyoma was made (Fig. 2B). At 5-year follow up, the patient is doing well and has had no recurrence (Fig. 2C). This case is presented to raise awareness of leiomyomas, although it is extremely rare in the periorbital region and difficult to distinguish. The histopathological and immunohistochemical features are essential for diagnosis. While complete excision is the usual outcome, long-term follow up is strongly recommended.