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Background: To describe a case of corneal toxicity associated with the use of the epidermal growth factor receptor (EGFR) inhibitor ABT-414 in the treatment of glioblastoma multiforme. Case Presentation: Case report of a 56-year-old male with glioblastoma multiforme who developed mild painless blurred vision after systemic treatment with the investigational EGFR inhibitor ABT-414. The patient had best corrected visual acuity of 20/60 right eye and 20/50 left eye. Ophthalmic examination revealed corneal toxicity with whorl-like opacities in the inferior interpalpebral cornea. The patient was treated with topical fluoromethalone and lifitegrast, and his ocular symptoms as well as the corneal findings improved. Conclusions: The systemic use of EGFR inhibitor ABT-414 may be associated with corneal toxicity and the effects are reversible with treatment.