Abstract Acute leukemia is a hematological malignancy affecting different organ systems including the eye and orbit through direct infiltration of tissues or secondary to hematological abnormalities. Ophthalmological manifestations in acute leukemia are variable ranging from asymptomatic presentation to serious manifestations that can alter the disease course and treatment. The purpose of this study is to detect the incidence of different ophthalmological manifestations in newly diagnosed acute leukemia patients and to assess the relationship between ocular findings and hematological characteristics and the sequel of these neoplasms. A cross-sectional study with analytical components was conducted on 222 newly diagnosed acute myeloid and acute lymphoblastic leukemia patients who presented at Oncology Center Mansoura University (OCMU) between January 2022 and February 2023. All patients underwent a complete ophthalmic evaluation at Mansoura Ophthalmology Center (MOC). The mean age was 43.45 ± 17.35 years (range, 17–85), and M/F was 137 (61.7%)/85 (38.3%). One-hundred and forty-four (64.9%) had acute myeloid leukemia (AML), and 78 (35.1%) had acute lymphoblastic leukemia (ALL). Ophthalmic manifestations were detected in 96 patients (43.2%). Among them, 4 (1.8%) had poor visual acuity. Retinal hemorrhage (19.8%) and Roth spots (17.1%) were the most common ocular manifestations. Other ophthalmological manifestations observed were orbital involvement (3.2%), ocular motility issues (1.4%), subconjunctival hemorrhage (5.9%), conjunctival chemosis (0.9%),lid swelling (4.1%), lid ecchymosis (3.2%), lagophthalmos (0.5%), lid ptosis (1.8%), retinal venous congestion & tortuosity (4.1%), preretinal hemorrhage (3.2%), vitreous hemorrhage (3.2%), macular affection (2.3%), retinal infiltration (1.8%), exudative retinal detachment (ERD) (1.8%), cotton-wool spots (0.9%), retinal vein occlusion (0.5%), papilledema (2.8%), optic disc infiltration (1.8%), disc pallor (1.8%).AML patients were significantly associated with a higher frequency of ocular affection, retinal hemorrhages, and Roth spots ( P 0.028, 0.003, and 0.046, respectively) compared to ALL patients. Retinal hemorrhage was statistically significantly associated with anemia ( P 0.021). Ophthalmological manifestations of acute leukemia are heterogeneous; they can be detected at initial presentations or relapse. Some manifestations are asymptomatic, others can affect visual acuity or even alter the disease course. Cooperation between ophthalmologists and haemato-oncologists is crucial for recognizing ocular involvement and disease management.