Objective and Importance: Pineal region tumors account for 0.4% to 1.0% of all intracranial tumors, but gliomas arising in the pineal region are rare; especially, oligodendroglioma arising in the pineal gland region is extremely rare, and only 4 cases have been reported so far. Clinical Presentation: A 23-year-old man developed headache and vomiting since June 20, 2010, and pineal region tumor and acute hydrocephalus were diagnosed. Magnetic resonance imaging on admission demonstrated heterogeneous, irregularly enhancing tumor in the region of the pineal gland, with compression of the cerebral aqueduct and hydrocephalus. Ventricular tap via the right anterior horn of the lateral ventricle, biopsy of the tumor in the pineal region by endoscopic approach, and third ventriculostomy for hydrocephalus were performed. Histopathological study and genetic evaluation of the pineal region tumor revealed anaplastic oligodendroglioma, and 4 courses of chemotherapy using carboplatin and etoposide were performed. Although the patient underwent chemotherapy, progression of multiple cerebrospinal fluid dissemination was observed. Conclusions: We have reported a very rare case of pineal region anaplastic oligodendroglioma. Even though there are extremely low incidences of pineal oligodendrogliomas, we should be alert for this pathology. Malignant oligodendroglioma is a risk factor for cerebrospinal fluid dissemination.
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