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一名患有癫痫和戈林-戈尔茨综合征的年轻患者的心脏性猝死未遂病例

Aborted sudden cardiac death in a young patient with epilepsy and the Gorlin Goltz syndrome.

作者信息

Salazar Serrano Guilherme, Dias de Oliveira Alexandre, Miotto Ramsés, Lin Katia, Loureiro Fialho Guilherme

机构信息

Department of Internal Medicine, University Hospital (HU), Universidade Federal de Santa Catarina (UFSC), Florianópolis, SC, Brazil.

Cardiology Division, Department of Internal Medicine, HU, UFSC, Florianópolis, SC, Brazil.

出版信息

Epilepsy Behav Rep. 2024 Apr 18;26:100667. doi: 10.1016/j.ebr.2024.100667. eCollection 2024.

DOI:10.1016/j.ebr.2024.100667
PMID:38699063
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11063993/
Abstract

Epilepsy is one of the most common chronical neurological conditions affecting over 50 million people worldwide. In addition to the stigma and discrimination, individuals with epilepsy suffer from a nearly three-fold increased risk of premature death compared to the general population. Although these premature deaths occur due to multiple causes, sudden unexpected death in epilepsy (SUDEP) still challenges neurologists and clinicians dealing with individuals with epilepsy. Recently, an increased interest in cardiac outcomes related to acute seizures and chronic epilepsy resulted in the groundbreaking development of the "epileptic heart" concept, and sudden death in individuals with epilepsy, which is 4.5 times as frequent as SUDEP according to some observational data, has gained more attention. As we gather information and learn about possible comorbidities and consequences of seizures and/or chronic epilepsy, we present a clinical case of a young patient with an unusual association of epilepsy, the Gorlin Goltz syndrome, and a cardiac fibroma with Wolf-Parkinson-White (WPW), who had multiple aborted cardiac arrests. Diagnostic challenges and multiple possible causes of sudden cardiac death in this single patient report are discussed.

摘要

癫痫是全球最常见的慢性神经系统疾病之一,影响着超过5000万人。除了污名化和歧视外,癫痫患者过早死亡的风险比普通人群高出近三倍。尽管这些过早死亡是由多种原因导致的,但癫痫猝死(SUDEP)仍然是困扰处理癫痫患者的神经科医生和临床医生的难题。最近,人们对与急性发作和慢性癫痫相关的心脏结局越来越感兴趣,这促成了“癫痫性心脏”概念的突破性发展,并且癫痫患者的猝死(根据一些观察数据,其发生率是SUDEP的4.5倍)受到了更多关注。在我们收集信息并了解癫痫发作和/或慢性癫痫可能的合并症及后果时,我们呈现了一名年轻患者的临床病例,该患者患有癫痫、戈林-戈尔茨综合征以及伴有预激综合征(WPW)的心脏纤维瘤,且多次发生心脏骤停未遂。本文讨论了该单一患者报告中诊断面临的挑战以及心脏性猝死的多种可能原因。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7f94/11063993/6f5c6f266059/gr4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7f94/11063993/7368e3728a1a/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7f94/11063993/f1cf5e356799/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7f94/11063993/541adb0da041/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7f94/11063993/6f5c6f266059/gr4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7f94/11063993/7368e3728a1a/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7f94/11063993/f1cf5e356799/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7f94/11063993/541adb0da041/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7f94/11063993/6f5c6f266059/gr4.jpg

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本文引用的文献

1
Epilepsy and the heart: can 'brain arrhythmia' lead to cardiac arrhythmias? Back to the basics.癫痫与心脏:“脑性心律失常”会引发心脏心律失常吗?回归基础。
Eur Heart J. 2024 Mar 7;45(10):853-854. doi: 10.1093/eurheartj/ehad807.
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Epilepsy and long-term risk of arrhythmias.癫痫与心律失常的长期风险。
Eur Heart J. 2023 Sep 14;44(35):3374-3382. doi: 10.1093/eurheartj/ehad523.
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Individuals with chronic epilepsy have elevated P-wave heterogeneity comparable to patients with atrial fibrillation.慢性癫痫患者的 P 波离散度增加,与房颤患者相当。
Epilepsia. 2023 Sep;64(9):2361-2372. doi: 10.1111/epi.17686. Epub 2023 Jun 28.
4
Seizures and status epilepticus may be risk factor for cardiac arrhythmia or cardiac arrest across multiple time frames.癫痫发作和癫痫持续状态可能是跨多个时间框架的心律失常或心搏骤停的危险因素。
Epilepsy Behav. 2021 Jul;120:107998. doi: 10.1016/j.yebeh.2021.107998. Epub 2021 May 12.
5
Ictal and Interictal Cardiac Manifestations in Epilepsy. A Review of Their Relation With an Altered Central Control of Autonomic Functions and With the Risk of SUDEP.癫痫发作期和发作间期的心脏表现。关于其与自主神经功能中枢控制改变及癫痫性猝死风险关系的综述
Front Neurol. 2021 Mar 12;12:642645. doi: 10.3389/fneur.2021.642645. eCollection 2021.
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Identifying patients with epilepsy at high risk of cardiac death: signs, risk factors and initial management of high risk of cardiac death.识别具有心脏性猝死高风险的癫痫患者:高危心脏性猝死的征象、危险因素和初始处理。
Epileptic Disord. 2021 Feb 1;23(1):17-39. doi: 10.1684/epd.2021.1254.
7
Risk of Cardiac Morbidities and Sudden Death in Patients With Epilepsy and No History of Cardiac Disease: A Population-Based Nationwide Study.患有癫痫且无心脏病史的患者发生心脏疾病和猝死的风险:一项基于人群的全国性研究。
Mayo Clin Proc. 2021 Apr;96(4):964-974. doi: 10.1016/j.mayocp.2020.04.050. Epub 2021 Jan 29.
8
Gorlin-Goltz syndrome and epilepsy: A two-case report and review of the literature.戈林-戈尔茨综合征与癫痫:两例报告及文献综述
Epilepsy Behav Rep. 2020 Aug 7;14:100384. doi: 10.1016/j.ebr.2020.100384. eCollection 2020.
9
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The Epileptic Heart: Concept and clinical evidence.癫痫心脏:概念与临床证据。
Epilepsy Behav. 2020 Apr;105:106946. doi: 10.1016/j.yebeh.2020.106946. Epub 2020 Feb 25.