Iampreechakul Prasert, Wangtanaphat Korrapakc, Wattanasen Yodkhwan, Hangsapruek Sunisa, Lertbutsayanukul Punjama, Siriwimonmas Somkiet
Department of Neurosurgery, Neurological Institute of Thailand, Bangkok, Thailand.
Department of Neuroradiology, Neurological Institute of Thailand, Bangkok, Thailand.
Surg Neurol Int. 2024 Jun 21;15:206. doi: 10.25259/SNI_45_2024. eCollection 2024.
Spontaneous obliteration of untreated cerebral arteriovenous malformations (AVMs) is rare, occurring in <1% of cases, and is even less common in pediatric populations. The mechanisms driving spontaneous regression of brain AVMs remain poorly understood, and long-term surveillance in pediatric patients is infrequently documented.
The authors reported a remarkably rare instance of spontaneous thrombosis in a pial AVM accompanied by a large intranidal aneurysm in a 10-month-old infant, initially presenting with a nocturnal seizure. Diagnostic imaging revealed a ruptured intranidal aneurysm causing acute hemorrhage in the left anterior interhemispheric subdural space, extending into adjacent areas. Further, magnetic resonance imaging (MRI) and magnetic resonance angiography delineated the AVM in the left superior frontal gyrus, associated with a thrombosed aneurysm and surrounding edema. Cerebral angiography confirmed the AVM's origin from the left anterior cerebral artery, displaying early venous drainage and small, indirect feeders not amenable to endovascular treatment. Over time, serial imaging showed the aneurysm's transition from partial to complete thrombosis. Subsequent MRIs and angiographic assessments up to age 10 confirmed complete resolution of the AVM and aneurysm, with focal hyperemia persisted until age 16, when recurrent AVM was identified.
We document a rare spontaneous regression of a pial AVM with an intranidal aneurysm influenced by specific vascular factors. Despite this, spontaneous thrombosis should not replace vigilant long-term monitoring in pediatric neurovascular care.
未经治疗的脑动静脉畸形（AVM）自然闭塞很少见，发生率<1%，在儿科人群中更为罕见。脑AVM自然消退的机制仍知之甚少，儿科患者的长期监测也鲜有记录。
作者报告了1例10个月大婴儿罕见的软膜AVM伴巨大瘤内动脉瘤自发血栓形成病例，最初表现为夜间癫痫发作。诊断性影像学检查显示瘤内动脉瘤破裂，导致左前半球间硬膜下间隙急性出血，并延伸至相邻区域。此外，磁共振成像（MRI）和磁共振血管造影显示左上额回的AVM，伴有血栓形成的动脉瘤和周围水肿。脑血管造影证实AVM起源于左大脑前动脉，显示早期静脉引流和不适合血管内治疗的小的间接供血动脉。随着时间的推移，系列影像学检查显示动脉瘤从部分血栓形成转变为完全血栓形成。后续直至10岁的MRI和血管造影评估证实AVM和动脉瘤完全消退，局灶性充血持续至16岁，此时发现复发性AVM。
我们记录了1例罕见的受特定血管因素影响的伴瘤内动脉瘤的软膜AVM自发消退病例。尽管如此，在儿科神经血管护理中，自发血栓形成不应取代警惕的长期监测。
