A 78-year-old man who had had uneventful extracapsular cataract extraction in the left eye 3 months earlier developed pyoderma gangrenosum (PG)-associated peripheral ulcerative keratitis (PUK) after suture removal. The patient had a 13-year history of PG associated with monoclonal immunoglobulin-A gammopathy. He presented with extensive, painful PUK at the incision site, with a descemetocele and a high risk for perforation. Fibrin glue tissue adhesive was used to stabilize the corneal ulcer as an adjunct to topical and systemic treatment. The patient had been treated with tapering doses of prednisone and cyclophosphamide (50 mg/day). High-dose human intravenous immunoglobulin (0.4 mg/kg/d for 4 days) was administered. The ulcer healed 1 month later with a loss of visual acuity. To our knowledge, this is the first reported case of PG-associated sclerokeratitis following cataract surgery. Early recognition of this rare ocular localization of PG is important to institute the appropriate therapy.