Gangliogliomas are mixed tumors which contain both glial and neuronal elements. The optic pathway is a very rare location for gangliogliomas, with less than 23 cases reported in the literature. Bilateral involvement of the entire optic pathway was reported in only 4 cases before. Because of similar radiological appearance of other pathological entities such as gliomas and craniopharyngiomas, histopathological diagnosis is essential. We report a ganglioglioma case that involved both optic pathways. A 12-year-old patient suffering from visual deterioration for 6 months was evaluated. After a visual field test and radiological examinations, a microsurgical biopsy procedure was performed. Pathological examination revealed dysplastic/neoplastic ganglion cells and neoplastic glial cells, and the diagnosis was a World Health Organization (WHO) grade 1 ganglioglioma. The patient is scheduled for adjuvant radiotherapy with the hope of prevention of progression.
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