Aim: Infliximab, an anti-tumor necrosis factor (TNF)-α monoclonal antibody, has been reported to be effective in refractory uveoretinitis in Behçet’s disease. Because it has been used clinically for a short time, information on its adverse effects is limited. We report a patient who developed cystoid macular edema (CME) following infliximab use for uveoretinitis associated with Behçet’s disease. Case Report: A 27-year-old man had refractory uveoretinitis and neuro-Behçet’s disease, and intravenous infliximab was administered. Results: One day after infliximab infusion, the patient complained of a decrease in the vision in his left eye. The visual acuity had decreased from 1.2 to 0.5. Daily optical coherence tomographic evaluations showed a progressive worsening of the CME, and fluorescein angiography showed a typical staining with a cystic pattern. Two weeks later, the height of CME appeared to reach a maximum level and thereafter gradually resolved in spite of the continuation of infliximab administration. The visual acuity improved while the patient was treated with repeated subtenon injections of steroids in addition to continuation of infliximab and finally increased from 0.15 to 1.2. Conclusions: Although the mechanism of CME is not known, clinicians should be aware that infliximab therapy might cause a development and worsening of CME. Thus, it is crucial to rule out preexisting abnormalities in the macula prior to commencing infliximab infusion.

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