Cystoid Macular Edema: Possible Complication of Infliximab Therapy in Behçet's Disease

Abstract

AIM: Infliximab, an anti-tumor necrosis factor (TNF)-alpha 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.

Fig. 1

Optical coherence tomographic images of the left eye before and after the infliximab injection. a No obvious abnormalities are seen in the left macula 1 day before the first infliximab infusion. Visual acuity was 1.2. b One day after the first infliximab infusion, the outer nuclear layer appears to be thicker at the macula suggesting the beginning of macular edema. Visual acuity was 0.5. c Two days after the first infliximab infusion, macular edema can be seen. Visual acuity was 0.1. d Four days after the first infliximab infusion, OCT image showing that the macular edema is worsened and cystoid spaces in the inner nuclear layer can also be seen. This image was obtained on the same day as the fundus photographs of figures 2c and d. Despite the OCT findings, visual acuity improved to 0.6. e Fourteen days after the first infliximab infusion, localized serous retinal detachment developed within the still remaining macular edema. Again, contrary to the OCT findings, visual acuity further improved to 0.7. f One month after the first infliximab infusion, the macular edema is resolved and visual acuity improved to 1.2.

Fig. 2

Fundus FA before and after infliximab infusion. Top: FA, performed 1 day before the first infusion of infliximab, showed diffuse dye leakage involving macula of the right eye (a) and slight macular leakage and retinal vasculitis in the left eye (b). Bottom: FA, performed 4 days after the first infusion of infliximab (taken on the same day as that in fig. 1d), shows still active diffuse dye leakage involving the macula in the right eye (c) and extensive dye pooling and staining with a cystic pattern in the macula in the left eye (d).