Endometriosis afflicts 10% of women in their reproductive years and nearly half of women with infertility and its etiology is not yet clear. Pharmacological therapy is generally based on progestins like progestogen. This drug binds to progesterone receptors with many known side effects. Here we described the case of a 33-year-old woman surgically treated for endometriosis continued with drug therapy based on estradiol valerate and dienogest. Approximately 21 months after treatment, she reported ocular symptoms with vision alteration, diplopia and metamorphopsia related to central serous chorioretinopathy (CSC). After discontinuation of combined progestin-based treatment, CSC fully subsided. Semeiological, clinical and laboratory approaches were adopted; urinary steroids were measured. A slight increase in prolactinemia in the absence of macro-prolactinemia was reported. Steroidal profile appeared without abnormalities, without abnormalities, although a slight alteration of estrogens balance was noted. Considering the pharmacodynamics of dienogest versus selective progesterone receptor modulators, it can be assumed that patients clinical events are related to specific-site-response of steroids that bind the progesterone receptor. Dienogest may have induced the CSC, as a not yet characterized side effect of the drug. Undoubtedly, further specific studies are needed concerning the metabolic and pharmacodynamic aspects that cannot be exhaustively covered here.