Shen Ying-Chi, Wu Jen-Chueh, Lin Ting-Tse, Chang Kai-Chung, Su Jen-Jen, Juang Jyh-Ming Jimmy
Department of Neurology, National Taiwan University Hospital, Taipei, Taiwan.
Department of Internal Medicine, National Taiwan University Hospital, Taipei, Taiwan.
Front Cardiovasc Med. 2024 May 31;11:1406614. doi: 10.3389/fcvm.2024.1406614. eCollection 2024.
Lacosamide is frequently used as a mono- or adjunctive therapy for the treatment of adults with epilepsy. Although lacosamide is known to act on both neuronal and cardiac sodium channels, potentially leading to cardiac arrhythmias, including Brugada syndrome (BrS), its adverse effects in individuals with genetic susceptibility are less understood.
We report a 33-year-old female with underlying epilepsy who presented to the emergency department with a four-day history of seizure clusters, and was initially treated with lacosamide therapy. During the intravenous lacosamide infusion, the patient developed sudden cardiac arrest caused by ventricular arrhythmias necessitating resuscitation. Of note, the patient had a family history of sudden cardiac death. Workup including routine laboratory results, 12-lead electrocardiogram (ECG), echocardiogram, and coronary angiogram was non-specific. However, a characteristic type 1 Brugada ECG pattern was identified by ajmaline provocation testing; thus, confirming the diagnosis of BrS. Subsequently, the genotypic diagnosis was confirmed by Sanger sequencing, which revealed a heterozygous mutation (c.2893C>T, p.Arg965Cys) in the gene. Eventually, the patient underwent implantable cardioverter-defibrillator implantation and was discharged with full neurological recovery.
This case highlights a rare but lethal adverse event associated with lacosamide treatment in patients with genetic susceptibility. Further research is warranted to investigate the interactions between lacosamide and variants.
拉科酰胺常用于治疗成人癫痫,作为单一疗法或辅助疗法。尽管已知拉科酰胺作用于神经元和心脏钠通道,可能导致心律失常,包括 Brugada 综合征(BrS),但其在遗传易感性个体中的不良反应尚鲜为人知。
我们报告一名 33 岁患有潜在癫痫的女性,因癫痫持续发作 4 天就诊于急诊科,最初接受拉科酰胺治疗。在静脉输注拉科酰胺期间,患者因室性心律失常发生心脏骤停,需要进行复苏。值得注意的是,该患者有心脏性猝死家族史。包括常规实验室检查结果、12 导联心电图(ECG)、超声心动图和冠状动脉造影在内的检查均无特异性。然而,通过阿义马林激发试验确定了特征性的 1 型 Brugada 心电图模式;从而确诊为 BrS。随后,通过桑格测序证实了基因诊断,结果显示该基因存在杂合突变(c.2893C>T,p.Arg965Cys)。最终,患者接受了植入式心律转复除颤器植入术,出院时神经功能完全恢复。
本病例突出了拉科酰胺治疗在遗传易感性患者中罕见但致命的不良事件。有必要进一步研究以探讨拉科酰胺与基因变异之间的相互作用。