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Jihene Sayadi,1 Dhouha Gouider,1 Yasmine Allouche,1 Racem Choura,1 Ines Cherni,1 Malek Sayadi,2 Hend Benneji,2 Imene Zghal,1 Ines Malek,1 Leila Nacef1 1Department A, Hedi Raies Institute of Ophthalmology, Faculty of Medicine of Tunis, University of Tunis El Manar, Tunis, Tunisia; 2Department of Hematology, Aziza Othmana Hospital, Faculty of Medicine of Tunis, University of Tunis El Manar, Tunis, TunisiaCorrespondence: Dhouha Gouider, Department A, Hedi Raies Institute of Ophthalmology, Faculty of Medicine of Tunis, University of Tunis El Manar, Bab Saadoun, Tunis, Tunisia, Tel +21640660114, Email dhouhagouider@gmail.comPurpose: 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/sub-hyaloid 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.Keywords: acute leukemia, ocular manifestations, leukemic retinopathy, retinal hemorrhage