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遗传性难治性癫痫患者的癫痫手术:一项系统评价

Epilepsy surgery for patients with genetic refractory epilepsy: a systematic review.

作者信息

Stevelink Remi, Sanders Maurits Wcb, Tuinman Maarten P, Brilstra Eva H, Koeleman Bobby Pc, Jansen Floor E, Braun Kees Pj

机构信息

Department of Child Neurology, Brain Center Rudolf Magnus, University Medical Center Utrecht.

Department of Genetics, Center for Molecular Medicine, University Medical Center Utrecht, The Netherlands.

出版信息

Epileptic Disord. 2018 Apr 1;20(2):99-115. doi: 10.1684/epd.2018.0959.

Abstract

In recent years, many different DNA mutations underlying the development of refractory epilepsy have been discovered. However, genetic diagnostics are still not routinely performed during presurgical evaluation and reports on epilepsy surgery outcome for patients with genetic refractory epilepsy are limited. We aimed to create an overview of the literature on seizure outcome following epilepsy surgery in patients with different genetic causes of refractory epilepsy. We systematically searched PubMed and Embase prior to January 2017 and included studies describing treatment outcome following epilepsy surgery in patients with genetic causes of epilepsy. We excluded studies in which patients were described with epilepsy due to Tuberous Sclerosis Complex or Sturge-Weber syndrome (since this extensive body of research has recently been described elsewhere) and articles in which surgery was aimed to be palliative. We identified 24 eligible articles, comprising a total of 82 patients who had undergone surgery for (mainly childhood-onset) refractory epilepsy due to 15 different underlying genetic causes. The success rate of surgery varied widely across these different genetic causes. Surgery was almost never effective in patients with epilepsy due to mutations in genes involved in channel function and synaptic transmission, whereas surgery was significantly more successful regarding seizure control in patients with epilepsy due to mutations in the mTOR pathway. Patients with a lesion on MRI tended to have higher seizure freedom rates than those who were MRI-negative. Although the evidence is still scarce, this systematic review suggests that studying genetic variations in patients with refractory epilepsy could help guide the selection of surgical candidates.

摘要

近年来,已经发现了许多导致难治性癫痫发生的不同DNA突变。然而,在术前评估期间仍未常规进行基因诊断,并且关于遗传性难治性癫痫患者癫痫手术结果的报告有限。我们旨在对不同遗传原因导致的难治性癫痫患者癫痫手术后发作结果的文献进行综述。我们在2017年1月之前系统地检索了PubMed和Embase,并纳入了描述遗传性癫痫患者癫痫手术后治疗结果的研究。我们排除了将患有结节性硬化症或斯-韦综合征所致癫痫的患者纳入研究的文献(因为最近在其他地方已经描述了这一广泛的研究内容)以及旨在进行姑息性手术的文章。我们确定了24篇符合条件的文章,共包括82例因15种不同潜在遗传原因接受(主要是儿童期起病的)难治性癫痫手术的患者。在这些不同的遗传原因中,手术成功率差异很大。对于因参与通道功能和突触传递的基因突变而导致癫痫的患者,手术几乎无效,而对于因mTOR通路突变而导致癫痫的患者,手术在控制发作方面明显更成功。MRI有病变的患者癫痫发作自由度往往高于MRI阴性的患者。尽管证据仍然很少,但这项系统综述表明,研究难治性癫痫患者的基因变异可能有助于指导手术候选者的选择。

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