Sickle cell disease is known to cause painful vaso-occlusive crises in long bones with large marrows. Orbital infarction is a rare complication of sickle cell disease and usually presents in children and adolescents with acute onset periocular swelling mimicking orbital cellulitis. We describe an atypical case of a 38-year-old man with homozygous sickle cell disease who presented with isolated, complete ptosis of his OD with minimal swelling. He described brow aches on the same side, with no features of proptosis, ophthalmoplegia, or compressive optic neuropathy. Orbital imaging revealed infarction in the right frontal bone and orbital roof, with inflammatory soft tissue swelling. His condition resolved completely after 4 days with conservative medical therapy alone.