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儿童海绵状血管畸形相关癫痫的手术治疗:病例系列、系统评价和荟萃分析。

Surgical treatment of cavernous malformation-related epilepsy in children: case series, systematic review, and meta-analysis.

机构信息

Child Neuropsychiatry, Department of Neuroscience, Rehabilitation, Ophthalmology, Genetics, Maternal and Child Health Department of Neuroscience (DINOGMI), IRCCS Istituto Giannina Gaslini, University of Genoa, Genoa, Italy.

Child Neuropsychiatry Unit, IRCCS Istituto Giannina Gaslini, Genoa, Italy.

出版信息

Neurosurg Rev. 2024 May 31;47(1):251. doi: 10.1007/s10143-024-02491-0.

Abstract

OBJECTIVE

Cerebral cavernous malformations (CCMs) are cerebral vascular lesions that occasionally occur with seizures. We present a retrospective case series from IRCCS Gaslini Children's Hospital, a systematic review, and meta-analysis of the literature with the goal of elucidating the post-surgery seizure outcome in children with CCMs.

METHODS

a retrospective review of children with cavernous malformation related epilepsy who underwent surgery at Gaslini Children's Hospital from 2005 to 2022 was conducted. We also conducted a comprehensive search on PubMed/MEDLINE and Scopus databases from January 1989 to August 2022. Inclusion criteria were: presence of CCMs-related epilepsy, in under 18 years old subjects with a clear lesion site. Presence of post-surgery seizure outcome and follow-up ≥ 12 months.

RESULTS

we identified 30 manuscripts and 223 patients with CCMs-related epilepsy, including 17 patients reported in our series. We identified 85.7% Engel class I subjects. The risk of expected neurological deficits was 3.7%; that of unexpected neurological deficits 2.8%. We found no statistically significant correlations between Engel class and the following factors: site of lesion, type of seizure, drug resistance, duration of disease, type of surgery, presence of multiple CCMs. However, we found some interesting trends: longer disease duration and drug resistance seem to be more frequent in subjects in Engel class II, III and IV; multiple cavernomas would not seem to influence seizure outcome.

CONCLUSIONS

epilepsy surgery in children with CCMs is a safe and successful treatment option. Further studies are necessary to define the impact of clinical features on seizure prognosis.

摘要

目的

脑海绵状血管畸形(CCMs)是一种偶尔伴有癫痫发作的脑血管病变。我们报道了来自 Gaslini 儿童医院的回顾性病例系列研究,对文献进行了系统评价和荟萃分析,旨在阐明 CCM 患儿手术后癫痫发作的结果。

方法

对 2005 年至 2022 年在 Gaslini 儿童医院因 CCM 相关癫痫接受手术的儿童进行回顾性研究。我们还对 1989 年 1 月至 2022 年 8 月期间 PubMed/MEDLINE 和 Scopus 数据库进行了全面检索。纳入标准为:存在 CCM 相关癫痫,患者年龄小于 18 岁,病变部位明确。存在术后癫痫发作结果和随访时间≥12 个月。

结果

我们确定了 30 篇文献和 223 例 CCM 相关癫痫患者,其中包括我们系列研究中的 17 例患者。我们发现 85.7%的患者属于 Engel Ⅰ级。预计出现神经功能缺损的风险为 3.7%,意外出现神经功能缺损的风险为 2.8%。我们发现 Engel 分级与以下因素之间无统计学显著相关性:病变部位、发作类型、耐药性、疾病持续时间、手术类型、多个 CCMs 的存在。然而,我们发现了一些有趣的趋势:Engel Ⅱ级、Ⅲ级和Ⅳ级患者的疾病持续时间和耐药性似乎更长;多发性海绵状血管畸形似乎不会影响癫痫发作的结果。

结论

对 CCM 儿童进行癫痫手术是一种安全且有效的治疗选择。需要进一步研究来确定临床特征对癫痫预后的影响。

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