Refractory De Novo Multiple Cerebral Aneurysms After Radiotherapy and Multistaged 'Open' Surgical Treatment for Low-Grade Glioma During Long-Term Follow-Up: A Case Report and Review of the Literature

Background: Radiation-induced aneurysms have been previously reported; however, multiple and repeated de novo aneurysm formation chronologically and anatomically during long-term follow-up have not yet been observed. The pathogenesis of persistent radiation-induced vasculopathy is not fully understood. Case Description: A 31-year-old woman presented with intraventricular hemorrhage due to rupture of a right internal carotid artery (ICA) aneurysm that developed 17 years after surgical resection of a low-grade glioma in the right frontal lobe and postoperative radiotherapy (focal, 50 Gy/25 fractions). During glioma follow-up, salvage surgery with adjuvant gamma knife therapy and chemotherapy (ranimustine, vincristine, temozolomide) were performed for recurrence of the glioma. The aneurysm was treated with endovascular coil embolization. However, she experienced repeated intraventricular hemorrhages, and angiography revealed a de novo ICA aneurysm. The de novo aneurysms were treated with endovascular surgery using coil embolization and stenting. At 2 years after the third hemorrhage, the surgical wound became dehiscent, probably due to wound infection, thus epicranial soft tissue reconstruction using vascularized skin flap was performed. Despite multistaged endovascular surgery for the ICA aneurysm, she experienced repeated subarachnoid and intraventricular hemorrhages. Angiography revealed a de novo aneurysm of the right posterior cerebral artery and basilar trunk. She underwent coil embolization and stenting. Despite active management with endovascular surgery and close follow-up, she died after an eighth consecutive intraventricular and intracerebral hemorrhage caused by a de novo large aneurysm of the posterior cerebral artery. Conclusions: To the best of our knowledge, the present study is the first to report on of refractory and recurring de novo aneurysms treated by multistaged endovascular surgery during a long-term follow-up after radiotherapy and multistaged craniotomy for glioma. Key words: De novo aneurysm, Low-grade glioma, Multistaged craniotomy, Occult wound infection, Radiation-induced aneurysm