Infliximab for the treatment of refractory progressive sterile peripheral ulcerative keratitis associated with late corneal perforation: 3-year follow-up.
OBJECTIVE
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.
Odorcic
Silvia
S
Faculty of Medicine, University of Toronto, Toronto, Canada.
Keystone
Edward C
EC
Ma
Joseph J K
JJ
D000368:Aged; D000893:Anti-Inflammatory Agents / Q000008:administration & dosage / Q000627:therapeutic use*; D000911:Antibodies, Monoclonal / Q000008:administration & dosage / Q000627:therapeutic use*; D002386:Cataract / Q000150:complications; D002387:Cataract Extraction / Q000379:methods; D003320:Corneal Ulcer / Q000150:complications / Q000188:drug therapy* / Q000473:pathology / Q000503:physiopathology; D018450:Disease Progression; D004305:Dose-Response Relationship, Drug; D004334:Drug Administration Schedule; D005500:Follow-Up Studies; D006801:Humans; D000069285:Infliximab; D007275:Injections, Intravenous; D007634:Keratitis / Q000150:complications / Q000188:drug therapy* / Q000503:physiopathology; D019654:Lens Implantation, Intraocular; D008297:Male; D018918:Phacoemulsification; D011241:Prednisone / Q000008:administration & dosage / Q000627:therapeutic use; D016896:Treatment Outcome; D014409:Tumor Necrosis Factor-alpha / Q000037:antagonists & inhibitors; D020246:Venous Thrombosis / Q000150:complications; D014792:Visual Acuity; D014945:Wound Healing
D000893:Anti-Inflammatory Agents; D000911:Antibodies, Monoclonal; D014409:Tumor Necrosis Factor-alpha; D000069285:Infliximab; D011241:Prednisone