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新生儿丘脑出血11年后诊断出的小儿脑动静脉畸形:病例说明

Pediatric cerebral arteriovenous malformation diagnosed 11 years after neonatal thalamic hemorrhage: illustrative case.

作者信息

Mochizuki Yoshiki, Maeda Takuma, Ooigawa Hidetoshi, Kobayashi Masahito, Kurita Hiroki

机构信息

Department of Cerebrovascular Surgery, Saitama Medical University International Medical Center, Hidaka, Japan.

Department of Neurosurgery, Saitama Medical University, Moro, Japan.

出版信息

J Neurosurg Case Lessons. 2025 May 5;9(18). doi: 10.3171/CASE25124.

Abstract

BACKGROUND

Arteriovenous malformations (AVMs) are known as congenital vascular disorders. However, ruptured AVMs in neonates are extremely rare. A hypothesis suggests that micro-AVM or de novo AVMs enlarge during childhood, eventually acquiring the pathological features of adult AVMs. The authors describe a case of a pediatric AVM diagnosed 11 years after neonatal intracerebral hemorrhage.

OBSERVATIONS

A 16-day-old neonate presented with sudden vomiting and convulsions. CT revealed a left thalamic hemorrhage extending into the left lateral ventricle. MRI showed no bleeding sources, including AVMs. By the age of 3 years, the patient developed West syndrome and was administered multiple antiseizure medication. At 5 years of age, the patient experienced convulsive status epilepticus, primarily on the right side of his body. A focal impaired awareness seizure due to the previous hemorrhage was considered. The patient remained seizure free until 11 years of age, when generalized seizures developed. MRI revealed a left thalamic AVM classified as Spetzler-Martin grade IV. The patient has been scheduled to undergo stereotactic radiosurgery.

LESSONS

Pediatric AVMs can undergo dynamic changes, including enlargement and de novo formation, acquiring the pathological features of adult AVMs. AVMs should be considered as a potential source of neonatal intracranial hemorrhage, necessitating long-term follow-up. https://thejns.org/doi/10.3171/CASE25124.

摘要

背景

动静脉畸形(AVM)是一种先天性血管疾病。然而,新生儿期破裂的AVM极为罕见。有一种假说认为,微小AVM或新生AVM在儿童期会增大,最终具备成人AVM的病理特征。作者报告了1例新生儿脑出血11年后确诊的儿童AVM病例。

观察结果

一名16日龄新生儿出现突发呕吐和惊厥。CT显示左侧丘脑出血并延伸至左侧侧脑室。MRI未发现包括AVM在内的出血源。患儿3岁时患韦斯特综合征,接受了多种抗癫痫药物治疗。5岁时,患儿出现惊厥性癫痫持续状态,主要累及身体右侧。考虑为既往出血导致的局灶性意识障碍性癫痫发作。该患儿在11岁前未再出现癫痫发作,11岁时出现全身性癫痫发作。MRI显示左侧丘脑AVM,分级为斯佩茨勒-马丁IV级。该患儿已安排接受立体定向放射外科治疗。

经验教训

儿童AVM可发生动态变化,包括增大和新生形成,具备成人AVM的病理特征。AVM应被视为新生儿颅内出血的潜在原因,需要长期随访。https://thejns.org/doi/10.3171/CASE25124

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8d19/12051992/61d18ea916b4/CASE25124_figure_1.jpg

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