Supratentorial ependymomas are rare, especially in the third ventricle. We report the case of an ependymoma of the posterior third ventricle that was endoscopically removed just by aspiration through a flexible scope. Histologically, beside the typical pattern of growth with perivascular pseudorosettes, the tumor featured hypercellular areas with more than 10 mitoses per 10 high-power fields, consistent with grade III-anaplastic tumor. A few months later, a second neuroendoscopy offered the unique chance to appreciate the total absence of tumor tissue and the restored anatomy. However, consistently with the high grade, the tumor recurred in two different locations including the endoscopic trajectory, and spread through the cerebrospinal fluid. The patient underwent a second resective surgery and radiosurgery. Despite a cycle of chemotherapy, multiple lesions both in the ventricular system and at the level of cauda equina appeared 12 months later. A comprehensive review of intraventricular anaplastic ependymomas is also provided.