Abstract

PURPOSE:

To report a case of refractory progressive sterile peripheral ulcerative keratitis (PUK) that resulted in late corneal perforation, despite good initial response to tumor necrosis factor-alpha inhibitor infliximab.

METHODS:

Review of the clinical course of a patient with progressive PUK treated with infliximab infusions of 3 mg/kg intravenously.

RESULTS:

A 72-year-old man presented with a visual acuity of 20/200 and a 5-month history of a progressive sterile PUK. More than 90% of the surface area previously unresponsive to 8 weeks of high-dose systemic steroid therapy healed within 1 week of the first infusion. After his second infusion, best-corrected visual acuity improved to 20/30(+2), with 2 small epithelial defects remaining. However, the remaining unhealed cornea thinned to an area of microperforation 6 weeks after his third dose, prompting an increase in dose frequency to every 4 weeks. One month after his fifth infusion, the area of ulceration healed completely. After his seventh infusion, the patient developed a deep venous thrombosis and infliximab was discontinued. After 10 months of remission, clear corneal cataract surgery was performed. Three years after initial presentation, he remains in remission with a corrected visual acuity of 20/20.

CONCLUSIONS:

Infliximab was effective in rapidly arresting the progression of a sterile PUK in our patient. Optimal dosing for infliximab in PUK has not been established, and increasing dose frequency to every 4 weeks may be necessary. Despite a progressive PUK resulting in corneal perforation, treatment with infliximab and subsequent visual rehabilitation can result in sustained remission and an excellent visual outcome.