Although rare, orbital bone marrow infarction is a serious complication in patients with sickle cell disease (SCD). Ophthalmologists should have a high index of suspicion for this disease in cases of acute childhood proptosis. Appropriate early management will prevent optic nerve dysfunction and permanent vision loss. Here, we present a case where the patient presented to the ophthalmologist with complaints of pain and proptosis, which led to the diagnosis of SCD.