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Background Optochiasmatic tuberculomas are very rare lesions. They can occur with concomitant tuberculous meningitis, and pulmonary tuberculosis or as the only manifestation of the disease. The authors present a case of optic pathways tuberculoma with radiologic appearance simulating an optic pathways glioma. Case description We report a case of a 20-year-old man with mental retardation due to anoxic encephalopathy who developed a sudden bilateral amaurosis. He also presented with diabetes insipidus, panhypopituitarism, right proptosis, and chemosis. Computed tomography (CT) and magnetic resonance imaging (MRI) showed an enhancing lesion in the optochiasmatic region extending to both optic nerves, with a mass in the right orbit, mimicking an optic pathways glioma. There was no other evidence of systemic involvement of the tuberculosis. The lesion was explored through a right pterional transylvian approach with opening of the optic canal and orbital roof, and a biopsy and an internal decompression were performed. Histopathological studies demonstrated a granulomatous lesion with central caseous necrosis with acid-fast bacilli. The patient improved after treatment with tuberculostatic drugs, but vision recovery could not be achieved. Conclusion Visual compromise in tuberculosis is associated with hydrocephalus, optical neuritis or tuberculomas involving the optic pathways. Reviewing the literature on tuberculomas of the optochiasmatic area, we could not find any other case with such extensive involvement of the optic pathways that was radiologically suggestive of an infiltrating glioma. Histopathological studies remain crucial in the diagnosis of intrinsic expansive processes of the optochiasmatic region.