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To present a case of central serous chorioretinopathy (CSC) in association with regressed familial retinoblastoma. A 23-year-old man with regressed unilateral familial retinoblastoma in his left eye presented with decreased vision of the left eye since two months ago. The patient had undergone chemotherapy and cryotherapy for the treatment of retinoblastoma 20 years ago. In the left eye, funduscopy disclosed regressed mass of retinoblastoma, inferonasal to the optic disc, and focal subfoveal neurosensory detachment. Optical coheren