Department of Neurosurgery, 5925The Mount Sinai Hospital, New York, NY, USA.
Neuroradiol J. 2022 Aug;35(4):533-538. doi: 10.1177/19714009211059115. Epub 2022 Jan 31.
Multiple de novo brain arteriovenous malformations (bAVM) have been reported in the literature, raising questions about the contended purely congenital nature of these lesions. We present the 15-year course of a pediatric patient, who initially presented at age 5 with a thalamic cavernous malformation and was treated with radiosurgery, and then subsequently developed a thalamic de novo bAVM immediately adjacent to the initial lesion location, discovered 2 years later. Treatment of the bAVM entailed two transarterial embolizations and one radiosurgery session which ultimately led to complete angiographic resolution. Finally, this patient's course was complicated by intraparenchymal hemorrhage and acute obstructive hydrocephalus, and further imaging revealed two newly formed cavernous malformations, also associated with the initial lesion's location, that have remained stable since their formation. This case likely represents the second-hit model for the formation of vascular malformations, as sparsely supported by the current literature. According to this, genetically aberrant, yet quiescent, brain areas might promote the de novo formation of vascular malformations after brain injury, including radiation.
已有文献报道多发性脑动静脉畸形(bAVM),这引发了这些病变是否具有纯粹先天性的争议。我们报告了一名儿科患者 15 年的病程,该患者最初在 5 岁时表现为丘脑海绵状畸形,并接受了放射外科治疗,随后在 2 年后发现紧邻初始病变部位的丘脑出现新的 bAVM。bAVM 的治疗包括两次经动脉栓塞和一次放射外科治疗,最终导致完全血管造影消退。最后,该患者的病程还伴有脑实质内出血和急性梗阻性脑积水,进一步的影像学检查显示两个新形成的海绵状畸形,也与初始病变部位有关,自形成以来一直保持稳定。根据目前文献的有限支持,这种情况可能代表了血管畸形形成的二次打击模型。根据这一模型,具有遗传异常但处于静止状态的脑区可能会在脑损伤(包括放射治疗)后促进新的血管畸形形成。