Jin Mei, Yang Fan, Du Yakun, Zhao Libo, Zhao Xueran, Liu Jing, Zhang Jing, Sun Suzhen
Department of Pediatric Neurology, The Children Hospital of Hebei Province, Shijiazhuang, Hebei, 050000, China.
The Key Laboratory of Pediatric Epilepsy and Neurological Disorders of Hebei Province, Shijiazhuang, Hebei, 050000, China.
BMC Pediatr. 2025 Mar 17;25(1):197. doi: 10.1186/s12887-025-05545-4.
A retrospective analysis was conducted to evaluate the clinical characteristics, diagnostic challenges, and management strategies in a child with congenital long QT syndrome (cLQTS) caused by a KCNH2 gene pathogenic variant presenting as "motor seizures". The case involved a 10-year-old boy with a two-year history of recurrent loss of consciousness, which had worsened during the preceding week. Clinical manifestations included sudden episodes of unconsciousness, rightward strabismus of both eyes, cyanosis of the lips, guttural vocalizations, rigidity and shaking of the upper limbs, and urinary incontinence. These events typically lasted approximately two minutes, initially occurring semiannually but escalating to daily episodes over the past week, affecting both awake and sleep states. Video electroencephalography (VEEG) showed generalized slow waves and low voltage activity, while electrocardiography (ECG) demonstrated QTc prolongation, paired, and multi-source ventricular ectopy preceding torsades de pointes. Genetic testing identified a pathogenic c.1697G > A mutation in the KCNH2 gene corroborating the clinical diagnosis of cLQTS. Following confirmation, the patient was initiated on long-term oral therapy with propranolol and nicorandil. Under this regimen, the patient was seizure-free for 7-month. For patients with seizures or seizure-like episodes, such as extremity movement or rigidity, it is necessary to perform an ECG examination. Additionally, dynamic ECG and electrolyte assessments should be conducted when necessary to minimize the risk of misdiagnosis and inappropriate treatment. When VEEG shows a "slow-flat-slow" pattern, differentiation from A-S syndrome caused by malignant arrhythmias is critical. Once cLQTS is diagnosed, it is imperative to initiate prompt and aggressive treatment to mitigate the risks of syncope and sudden cardiac death.
进行了一项回顾性分析,以评估一名因KCNH2基因致病变异导致“运动性癫痫发作”的先天性长QT综合征(cLQTS)患儿的临床特征、诊断挑战和管理策略。该病例为一名10岁男孩,有两年反复意识丧失病史,前一周病情加重。临床表现包括突然意识丧失、双眼向右斜视、嘴唇发绀、喉音、上肢僵硬和颤抖以及尿失禁。这些发作通常持续约两分钟,最初半年发作一次,但在过去一周内升级为每日发作,影响清醒和睡眠状态。视频脑电图(VEEG)显示广泛性慢波和低电压活动,而心电图(ECG)显示QTc延长、成对和多源性室性早搏,随后出现尖端扭转型室性心动过速。基因检测发现KCNH2基因存在致病性c.1697G>A突变,证实了cLQTS的临床诊断。确诊后,患者开始长期口服普萘洛尔和尼可地尔治疗。在此治疗方案下,患者7个月无癫痫发作。对于有癫痫发作或癫痫样发作(如肢体运动或僵硬)的患者,有必要进行心电图检查。此外,必要时应进行动态心电图和电解质评估,以尽量减少误诊和不适当治疗的风险。当VEEG显示“慢-平-慢”模式时,与恶性心律失常引起的A-S综合征进行鉴别至关重要。一旦诊断为cLQTS,必须立即开始积极治疗,以降低晕厥和心源性猝死的风险。