Purpose Acute macular neuroretinopathy (AMN) is a visual-deteriorating rare clinical entity with an uncertain etiology. We aimed to report a case of AMN and underlying disease of acute myeloid leukemia (AML). Case presentation A thirty-five-year-old female patient with bone marrow biopsy confirmed AML, and bicytopenia, under chemotherapy, complained of sudden paracentral visual field defect in her right eye was referred. Visual acuity was 20/20 in both eyes. Posterior segment evaluation

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... multiple Roth's spots. Optical coherence tomography (OCT) demonstrated hyper-reflectivity band, in the outer nuclear layer and outer plexiform layer, nasal to the fovea of the right eye, and hyperreflective patch in outer retina segmentation en-face OCT, suggestive of the diagnosis of AMN. Nine days after AMN diagnosis, dyspnea, malaise, and cough was initiated. Ground glass opacities in lung CT scan, beside reverse transcription polymerase chain reaction of severe acute respiratory syndrome coronavirus-2, was conclusive of coronavirus disease 2019 (COVID-19). The patient deceased after 6 days. Conclusion We report a rare case of AMN following AML. Our findings support the role of ischemia in the outer retina, of which AML may contributed to the pathophysiological process. The patient has deceased less than 2 weeks from AMN initiation.