PURPOSE: Chronic myeloid leukemia (CML) is a rare malignant myeloproliferative disorder characterized by the presence of the Philadelphia chromosome, a reciprocal translocation between chromosomes 9 and 22. Ocular manifestations in CML are infrequent but can be the initial indicators of the disease. We report the case of a 28-year-old male patient with suspected hyperviscosity syndrome secondary to CML, based on fundus findings and supported by multimodal imaging. METHODS: Multimodal imaging was performed to assess the ocular manifestations, before and after treatment. RESULTS: The patient presented with posterior segment findings consistent with hyperviscosity syndrome, including retinal hemorrhages, vascular dilation, and tortuosity as demonstrated by multimodal imaging techniques. Further investigations confirmed the diagnosis of CML, and treatment with hydroxyurea, allopurinol, and imatinib led to a significant improvement in visual acuity and resolution of posterior segment findings. CONCLUSION: Ocular manifestations in CML vary widely and include both anterior and posterior segment involvement. Regular ophthalmic examinations are essential for the early detection of relapses or complications. This case underscores the importance of interdisciplinary collaboration between ophthalmologists and hematologists for the timely diagnosis and management of ocular manifestations in CML.