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To report visualization of a case of retinal deep capillary plexus ischemia with antiphospholipid syndrome.A 53-year-old woman was referred with a 1-week history of sudden onset of decreased vision in the right eye. Her symptoms were evaluated by clinical examination, infrared reflectance, fundus autofluorescence, fluorescein angiography, spectral-domain optical coherence tomography, microperimetry, and multifocal electroretinography.The patient's visual acuity on presentation was 0.4 (Snellen) in the right eye and 0.7 in the left eye. Spectral-domain optical coherence tomography showed increased reflectivity of inner nuclear and inner plexiform layers of the right eye. Laboratory evaluation revealed abnormal titers of lupus anticoagulant antibodies. After 6 months, her visual acuity was finger counting at 1 m in the right eye and 0.4 in the left eye. Spectral-domain optical coherence tomographic image demonstrated a diffuse thinning of those retinal layers in the right eye. These findings are consistent with the clinical characteristics of retinal deep capillary ischemia in association with antiphospholipid syndrome.Deep capillary ischemia has some characteristic findings, and on spectral-domain optical coherence tomography, outcomes in conjunction with multimodal imaging are helpful for the diagnosis of the acute and the chronic stages of retinal deep capillary plexus ischemia.