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To evaluate the clinical efficacy of topical mitomycin C (MMC) 0.04% in the treatment of recurrent corneal intraepithelial neoplasia.A 71-year-old man presented with a limbal tumor that had extended onto the inferior temporal quadrant of the left cornea. Superficial lamellar sclerokeratectomy was performed to remove the tumor, which was histopathologically demonstrated to be a corneal intraepithelial neoplasia. Fifteen months after the surgical excision, the tumor recurred at the previous keratectomy site. The patient was treated topically with 0.04% MMC four times daily for 2 weeks, during which time he only had a mild degree of conjunctival hyperemia and ocular pain.The tumor resolved completely after 2 weeks of treatment and did not recur during the 10-month follow-up.Topical MMC may be an effective alternative treatment for recurrent corneal intraepithelial neoplasia and deserves further evaluation.