Patient: Male, 17Final Diagnosis: Burkitt lymphomaSymptoms: Anisocoria, ipsilateral ptosis, opthalmoparesis, paresisMedication: —Clinical Procedure: —Specialty: OncologyObjective:Unusual clinical courseBackground:Burkitt lymphoma rarely affects the central nervous system and ocular region. Under these conditions, computed tomography and (particularly) magnetic resonance imaging of the skull increase the diagnostic accuracy, as they objectively show the topography of lesions and the effect of neoplasia on structures.Case Report:We report here the case of a 17-year-old male whose initial clinical manifestations were related to neurological impairment and to the ocular musculature and ocular innervation. The diagnosis of Burkitt lymphoma with leukemization and infiltration of the central nervous system was confirmed.Conclusions:In this case, it is important to recognize that the neuroimaging findings were fundamentally important in indicating the initial form of the disease and in directing the appropriate clinical management.