Herein, we present an extremely rare and unique case of a septum pellucidum cavernoma that occurred in a young male who had previously undergone brain radiotherapy in childhood due to acute lymphoid leukemia. Following presentation, he was diagnosed with generalized seizures. The cavernoma was resected via an anterior interhemispheric transcallosal approach, following which the patient remained stable without neurological sequelae.

To conclude, ventricular cavernomas are rare lesions, especially when located at the septum pellucidum, thus constitute a challenging neurosurgical approach. Keywords : Cavernous hemangioma, Septum pellucidum, Radiotherapy, Cerebral ventricle neoplasm, Cerebral hemorrhage