Abstract
PURPOSE: To report a case of unilateral isolated optic nerve infiltration combined with central retinal artery occlusion in a patient with acute myeloid leukemia. OBSERVATIONS: A 28-year-old acute myeloid leukemia patient with systemic remission after hematopoietic stem cell transplantation presented with optic nerve infiltration combined with central retinal artery occlusion. Fundus examination showed diffuse papillary swelling with blurred margins and whitening macular with cherry-red spot. CBFβ-MYH11 fusion transcript analysis revealed central nervous system relapse of acute myeloid leukemia and optic nerve infiltration. CONCLUSIONS AND IMPORTANCE: Isolated optic nerve infiltration combined with central retinal artery occlusion is an extremely rare presentation in acute myeloid leukemia. Even in the absence of cytological and radiographic evidence from cerebrospinal fluid, optic nerve infiltration should not be ignored.