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To report a case of unilateral exudative retinal detachment as the sole presentation of relapsing B-type lymphoblastic leukemia in a 35-year-old man after 3 years of remission.Case report.A 35-year-old man in complete remission of high-risk type B acute lymphoblastic leukemia (ALL-B) presented with acute vision loss in his left eye. Exudative retinal detachment was diagnosed at initial evaluation. Hematological and ocular studies were performed. Although there was no evidence of blood, cerebrospinal fluid, or bone marrow disease relapse, transvitreal retinochoroidal cytology identified the infiltration of lymphoblastic leukemic B cells with t(12:21) translocation as the only sign of relapsing ALL-B.At first sign of vision loss or ocular symptoms in a patient with a history of lymphoblastic leukemia, and regardless of the hematological findings, exhaustive ophthalmologic exploration and ocular biopsy should be performed without delay—this being crucial for clinical decision making because a prompt diagnosis improves the chances of survival.