Mahajan Uma V, Patel Mohit, Pace Jonathan, Rothstein Brian D
Department of Neurosurgery, University Hospitals Cleveland Medical Center, and Case Western Reserve University School of Medicine, Cleveland, Ohio, USA.
Brain Circ. 2022 Sep 21;8(3):121-126. doi: 10.4103/bc.bc_26_22. eCollection 2022 Jul-Sep.
Cerebral cavernous malformations (CMs) are slow-flow vascular lesions that affect up to 0.5% of the pediatric population. These lesions are at risk for hemorrhage, causing seizures, and leading to neurological deficits. Here, we conduct a literature review and then present a report of a supratentorial CM in a 2-year-old patient with no significant past medical history who presented at our institution with 1 month of eye twitching. We performed a literature search of five databases of all articles published before 2020. Our inclusion criteria included cohort and case series of children with mean age under 12 years. Our search yielded 497 unique articles, of which 16 met our inclusion criteria. In our pooled literature analysis, a total of 558 children were included, 8.3% of which had a positive family history and 15.9% had multiple CMs. About 46.1% of the children had seizures, and 88.4% of those who underwent surgery had a total resection. About 85.1% of those with epilepsy were Engel Class 1 postsurgery. Over a mean follow-up of 4.1 years, 3.4% of patients had additional neurological deficits, including paresis and speech deficits. Our analysis of published literature shows surgical intervention should be considered first-line therapy for patients who are symptomatic from CM, present with seizure, and have surgically accessible lesions. Additional work is needed on outcomes and long-term effects of minimally invasive treatments, including radiosurgery and laser ablation, in pediatric populations.
脑海绵状血管畸形(CMs)是一种低流量血管病变,影响着高达0.5%的儿科人群。这些病变有出血风险,可导致癫痫发作,并引起神经功能缺损。在此,我们进行了一项文献综述,然后报告了一名2岁幕上CM患者的病例,该患者既往无重大病史,因1个月的眼部抽搐前来我院就诊。我们在五个数据库中检索了2020年以前发表的所有文章。我们的纳入标准包括平均年龄在12岁以下儿童的队列研究和病例系列。我们的检索产生了497篇独特的文章,其中16篇符合我们的纳入标准。在我们的汇总文献分析中,共纳入了558名儿童,其中8.3%有家族史阳性,15.9%有多发性CMs。约46.1%的儿童有癫痫发作,接受手术的儿童中有88.4%实现了全切。癫痫患者术后约85.1%为恩格尔1级。在平均4.1年的随访中,3.4%的患者出现了额外的神经功能缺损,包括轻瘫和言语缺陷。我们对已发表文献的分析表明,对于有CM症状、出现癫痫发作且病变可通过手术切除的患者,应考虑手术干预作为一线治疗。对于儿科人群中包括放射外科和激光消融在内的微创治疗的疗效和长期影响,还需要进一步研究。