To report a case of isolated conjunctival lymphangioma mimicking a recurrent conjunctival cyst.

A 39-year-old male with a conjunctival cyst in the right eye lasted for 1 month visited our hospital. He had previously undergone aspiration of the cyst at another hospital 1 week before visiting our hospital. However, the cyst recurred, and he was referred to our hospital. On slit lamp biomicroscopy, yellow-colored turbid fluid and a hemorrhage were observed in the conjunctival cyst, but no specific finding was found in the fundus photography. The patient was initially treated with topical antibiotics and steroids. Three weeks later, absorption of the hemorrhage was noted, but there was no change in the size of the cyst. Therefore, surgical removal and histological examination of the cyst were performed. The histological examination revealed that the lesion was positive for CD 31 and D2-40, and the cyst was diagnosed as a cystic conjunctival lymphangioma. Thereafter, brain magnetic resonance imaging was performed to screen for orbital lymphangioma and systemic disease that could accompany a conjunctival lymphangioma. However, no specific findings were observed. There was no recurrence of the conjunctival cyst at 1 year and 6 months after surgical removal, and no other ophthalmic or systemic complication was observed.

An isolated conjunctival lymphangioma is a rare lesion. In the diagnosis of lymphangioma, systemic examination is recommended for the identification of comorbid diseases, such as orbital lymphangioma.