Gangliogliomas represent a rare form of neuroepithelial tumours, which even more rarely present with hemorrhage or localize intraventricularly. To date, only two cases of ganglioglioma with both of these features have been reported. Our patient is a 23-year-old woman who presented with signs and symptoms of increased ICP, with a post-subtotal resection diagnosis of WHO Grade I ganglioglioma localizing bilaterally to the lateral ventricles. One year following the operation, the tumour showed radiologic evidence of interval hemorrhage, which was verified histopathologically following a second subtotal resection. Greater than 95% of the lesion represented a large hematoma with organization and well-defined fibrous pseudo-capsule, with very scanty fragments of adjacent/peripheral low-grade glial tumour. This case represents a very rare presentation of intraventricular ganglioglioma with hemorrhage.