Abstract. The current study reports a case of a patient with a chronic myelocytic leukemic fundus lesion, initially diagnosed in the Department of Ophthalmology, Rizhao People's Hospital (Rizhao, China). A male, 23-years-of-age, presented with a dark shadow in the front of the right eye, accompanied with blurred vision for 3 days (visual acuity of right eye, 0.4; visual acuity of left eye, 0.6). In addition, the patient had experienced gingival bleeding for 2 years, and recurrent upper respiratory infections for 1 year. A fundus examination revealed mild binocular papillary edema, dilated and tortuous veins, and the retina exhibited large quantities of scattered and dark red bleeding spots. The bleeding spots had white spots in the center, which exhibited typical Roth spots, whilst routine blood examination, abdominal ultrasound, marrow biopsy and other laboratory tests confirmed the diagnosis of chronic myelogenous leukemia. Patients with leukemia typically present with initial symptoms that include fever, fatigue, anemia and hepatosplenomegaly, and a diagnosis as a result of eye-related symptoms is rare, rendering the present case unique.