• Mariko Kawashima, Hirotaka Hasegawa, Hiroki Kurita, Kaima Suzuki, Masahiro Shin, Masako Ikemura, and Nobuhito Saito.
• Department of Neurosurgery, University of Tokyo Hospital, Tokyo, Japan.
• World Neurosurg. 2020 Mar 1; 135: 63-67.

BackgroundWhile prior studies reporting de novo arteriovenous malformations after birth are not uncommon, reports of ectopic recurrence of arteriovenous malformation after radiotherapy are scarce.Case DescriptionAn 8-year-old girl with a ruptured cerebellar arteriovenous malformation was treated with stereotactic radiosurgery. The nidus was completely obliterated 17 months after stereotactic radiosurgery; however, 20 years later she had a rupture of a recurrent nidus that occurred adjacent to the original nidus, accompanied by a radiation-induced cyst. Surgical resection was performed, and subsequent pathologic examination revealed 2 different compartments: a typical nidus and a postirradiated degenerated nidus with an encapsulated hematoma. Immunohistochemical stains showed increased expression of Ki-67 and decreased expression of endoglin in endothelial cells in the recurrent nidus.ConclusionsDepletion in endoglin may lead to the formation of vessel malformations in the presence of angiogenic stimuli. This case serves as a strong reminder of the importance of long-term follow-up after stereotactic radiosurgery, especially for pediatric cases.Copyright © 2019 Elsevier Inc. All rights reserved.

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