Purpose: To report a rare case of a simultaneous bilateral central retinal vein occlusion, as the initial presentation of chronic myeloid leukemia.

Case summary: A 22-year-old man presented with conjunctival injection of both eye, decreased visual acuity of left eye, nausea and dizziness. Ophthalmic examination was consistent with non-ischemic central retinal vein occlusion of both eye. After a systemic evaluation including laboratory examinations, bone marrow biopsy, and abdomen computed tomography, he was diagnosed as chronic myeloid leukemia. The patient received massive hydration and chemotherapy with hydroxyurea, allopurinol, and torsemide. On follow-up at 2 months, his visual acuity recovered to 0.8 in right eye, and 0.63 in left eye. The ocular status has remained stable at 2 year of follow-up.

Conclusions: All ocular structures have been found to be involved in patients with leukemia, but visual impairment as the initial symptom of leukemia is rare. A prompt search for the underlying systemic disease should be performed in young patients presenting with central retinal vein occlusion.