Ectopic Recurrence of Arteriovenous Malformation After Radiosurgery: Case Report and Insight Regarding Pathogenesis

World Neurosurg. 2020 Mar:135:63-67. doi: 10.1016/j.wneu.2019.11.156. Epub 2019 Dec 10.

Abstract

Background: While prior studies reporting de novo arteriovenous malformations after birth are not uncommon, reports of ectopic recurrence of arteriovenous malformation after radiotherapy are scarce.

Case description: An 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.

Conclusions: Depletion 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.

Keywords: Arteriovenous malformation; Endoglin; Gamma Knife; Stereotactic radiosurgery.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Brain Neoplasms / etiology
  • Brain Neoplasms / pathology
  • Brain Neoplasms / surgery
  • Female
  • Hematoma / etiology
  • Hematoma / pathology
  • Hematoma / surgery
  • Humans
  • Intracranial Arteriovenous Malformations / etiology*
  • Intracranial Arteriovenous Malformations / pathology
  • Intracranial Arteriovenous Malformations / therapy*
  • Neoplasms, Radiation-Induced / pathology
  • Neoplasms, Radiation-Induced / surgery
  • Radiosurgery*
  • Recurrence
  • Rupture, Spontaneous / etiology
  • Rupture, Spontaneous / pathology
  • Rupture, Spontaneous / radiotherapy