Subependymomas are rare brain tumors derived from the ventricular system and are usually asymptomatic. The interest of this report is to demonstrate that subependymomas can very exceptionally present bilaterally and with hemorrhage. A 69-year-old woman presented with headache, ataxia, and focal neurologic deficits. Magnetic resonance imaging revealed a bilateral intra-axial lesion with unilateral hemorrhage. Patient underwent surgical resection of the tumor and the pathology was consistent with subependymoma. Postoperatively, patient recovered progressively with improvement of symptoms. This is the first case of a subependymoma that is both bilateral and hemorrhagic, two rare occurrences previously mutually exclusive in literature for this type of tumor. In the present case report we perform a review of the literature to analyze and compare retrospectively all other cases of hemorrhagic subependymomas.
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