Antiphospholipid syndrome (APS) is a systemic autoimmune disorder characterized by arterial and/or venous thrombosis, recurrent fetal losses or other pregnancy complications, and the presence of antiphospholipid antibodies (aPL). Ocular manifestations occur in 8-88% of patients with APS and are typically due to vaso-occlusive disease involving retinal and choroidal vessels. We report an unusual case of neuroretinitis as a first presentation of lupus-like illness with APS.