Objective: Central neurocytomas are rare intraventricular tumors with neuronal differentiation, typically located in the ventricles while tumors located outside the ventricles designated as ''extraventricular neurocytomas'' (EVN). Neurocytomas are classified as atypical if they exhibit a MIB-1 labeling index >2 % or atypical features, like focal necrosis, vascular proliferation, and increased mitotic activity. We here report a case of atypical EVN of the left frontal lobe. Method: A 60-year-old woman patient admitted to hospital for headache. CT scan showed a large partially calcified cystic mass. MR imaging showed circumscribed, solid mural nodule within the cystic mass. The patient underwent a left frontal craniotomy. Specimens were fixed in 10 % formalin and stained with hematoxylin and eosin. For immunohistochemistry, sections were incubated with monoclonal antibodies to synaptophysin, neurofilament protein, MAP-2, NSE and GFAP. The proliferative index was assessed with MIB-1. Results: Histopathological examination revealed a neuronal neoplasm composed of uniform cells in solid sheets. Small necrosis areas and mitotic figures were evident. Immunohistochemical examination revealed positive results for synaptophysin, neurofilament protein, MAP-2, and NSE. GFAP staining was negative. MIB-1 index was calculated as 5 %.