Purpose: To report a patient with corneolimbal squamous dysplasia successfully treated with interferon therapy. Method: A 78-year-old man had a gelatinous and vascularized lesion involving the left cornea and adjacent limbus. Impression cytology studied with periodic acid—Schiff-hematoxylin stain disclosed severe squamous dysplasia. Topical instillation and subconjunctival injection of interferon alpha-2b was instituted. Results: The gelatinous tumor mass involving the cornea and adjacent limbus completely resolved 2 months after interferon treatment. He showed no sign of recurrence during the 10-month follow-up. Conclusion: Interferon therapy may be a therapeutic alternative for treating corneolimbal squamous dysplasia.