To describe ocular manifestations of acute leukemia in a Tunisian cohort and to assess the associations between ophthalmic findings and epidemiological, clinical, and biological features of the disease. Methods: A prospective study included patients newly diagnosed with acute leukemia referred to our clinics between January 2019 and July 2020. All patients underwent a complete ophthalmic evaluation and spectral-domain optical coherence tomography (SD-OCT) at presentation, then every two months during one year. We defined two groups: Group 1 included patients with leukemic ophthalmopathy and group 2 included patients with normal ophthalmic examination. Results: Forty-six patients were enrolled. The mean age of patients was 32.1±15.3 years. The sex ratio M/F was 1.55 (28 male patients and 18 females). Twenty-nine patients (63%) had acute myeloid leukemia (AML), and 17 (37%) had acute lymphoblastic leukemia (ALL). The average follow-up was 9.1 months (range: 3-12 months). We observed ophthalmic manifestations in 28 patients (61%). Among them, 17 (61%) had vision-threatening complications. The posterior segment was the most common site of ocular involvement (82% of group1). Primary leukemic infiltration (Disc edema, ptosis, exophthalmos) was present in 13 eyes (14.1%). Twenty-seven eyes (29.3%) had secondary involvement lesions (Subconjunctival hemorrhage, periorbital ecchymosis, retinal/subhyaloid hemorrhage, dilated/tortuous veins). Twenty-one eyes (22.8%) showed other ocular manifestations which etiopathogenesis is not yet fully understood (White-centred hemorrhages, cotton-wool spots, serous retinal detachment, hemorrhagic pigment epithelial detachment). Leukemic retinopathy was significantly more frequent in adults (23/39 and 1/7 in adult and pediatric groups, respectively; p=0.003). Patients suffering from AML were more likely to have secondary ocular involvement (20/29 and 7/17 in AML and ALL patients, respectively; p=0.047). Retinal hemorrhages were statistically associated with anemia and thrombocytopenia (p=0.041 and p=0.034; respectively). Conclusion: Leukemic ophthalmopathy seems to be frequent and may lead to severe visual impairment. An ophthalmic assessment complemented with SD-OCT has paramount importance in all newly diagnosed acute leukemic patients.