PURPOSE: To present a case of peripheral ulcerative keratitis (PUK) that progressed to corneal perforation in the setting of recently diagnosed temporal arteritis. METHODS: A 76-year-old man presented with a 2-week history of red eye and gradual loss of vision in the right eye. His medical history included recently diagnosed temporal arteritis without previous eye involvement. Clinical examination revealed severe peripheral corneal melting leading to corneal perforation. RESULTS: The patient was treated in the acute phase with topical dexamethasone drops and oral prednisolone. Perforation was sealed with cyanoacrylate glue. Azathioprine was also administered. Inflammation was resolved and his eye remains quiet. CONCLUSIONS: To our knowledge, this is the first reported case of PUK in the background of temporal arteritis.