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Angiocentric glioma (AG) is a rare central nervous system (CNS) neoplasm that was only recently recognized by the World Health Organization (WHO). AG occurs in a broad age range, shows no gender predilection, and arises superficially in the cerebrum, usually resulting in medically intractable seizures. Most cases are cured by surgical excision alone, consistent with a WHO grade I neoplasm. We report a case of an AG in the right frontal lobe of a 57-year-old female, emphasizing the cytologic and immunohistochemical features, including confirmation and comparison with the surgical specimen. To our knowledge, this is the first report detailing the cytology of AG, including demonstration of important diagnostic findings that were only appreciated in the cytologic preparations and not in the smears or the surgical specimen. We also compare and contrast AG to other entities in the differential diagnosis and include a review of the literature. Diagn. Cytopathol. 2010;38:452-456.