Purpose: Iridocorneo-endothelial (ICE) syndrome is known as a rare spectrum causing glaucoma, corneal and iris damages. Retinal complications are uncommon., Observations: We report the case of a middle-aged woman suffering from a Cogan-Reese Syndrome (CRS) with refractory ocular hypertension (OHT) who presented a cystoid macular edema (CME) during follow up., Conclusions and Importance: We suspect the CME to be inflammatory linked to the pathophysiological hypotheses of the CRS. The CME was successfully treated with topical nonsteroidal anti-inflammatory drugs (NSAID). No consensus is available on its duration. A recurrence happened when treatment was stopped, its reintroduction was successful., Competing Interests: The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper. The following authors have no financial disclosures:BH, HP, AE, ECH, DC., (© 2022 The Authors.)