To report a case of idiopathic sclerosing inflammatory tumor presenting as painful masses in the medial side of both upper eyelids.

A 26-year-old female presented with pain in both eyes at upward gaze and progressive medial side masses on bilateral upper eyelids 3-4 months prior. Orbit MRI showed an orbital benign tumor and partial excisional biopsy was performed for the larger right upper eyelid mass. The biopsy result was chronic inflammation with fibrosis. There was no change in the masses size after an oral steroid was prescribed for 11 days. At 6 weeks after the first operation, complete excisional biopsy was performed for the bilateral upper eyelid masses and idiopathic sclerosing inflammatory tumor was diagnosed. Intravenous steroid injections were administered twice with a 1-week interval postoperatively. After 6 months of follow-up, no recurrence was evident.

Idiopathic sclerosing inflammatory tumor usually involves the anterior, lateral, or apex of the unilateral orbit and does not invade the inferomedial side of the orbit and typically has a chronic course. We experienced a rare case of idiopathic sclerosing inflammatory tumor that involved the medial side of both upper eyelids which was cured by complete excision.