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Paroxysmal sympathetic hyperactivity and the later development of epilepsy in a chemotherapy-associated brain damage
Brain and Development ( IF 1.4 ) Pub Date : 2021-07-21 , DOI: 10.1016/j.braindev.2021.07.001
Ryoji Taira 1 , Kenichiro Yamamura 2 , Tomoko Maeda 3 , Ayumi Sakata 3 , Eriko Watanabe 3 , Takafumi Shimogawa 4 , Nobutaka Mukae 4 , Chizuru Ikeda 5 , Masahiro Migita 6 , Osamu Watanabe 7 , Yuhki Koga 2 , Yasunari Sakai 1 , Shouichi Ohga 1
Affiliation  

Background

Chemotherapy in childhood leukemia potentially induces brain lesions and neurological sequelae. Paroxysmal sympathetic hyperactivity (PSH) is known as a treatment-associated complication; however, the full clinical spectra of PSH remain to be elusive.

Case report

A 5-year-old girl was diagnosed of acute myeloid leukemia (AML) M5. After the intensification therapy, she developed recurrent symptoms of episodic tachycardia, hypertension and perspiration lasting for several hours per day. The low-frequency-high-frequency ratio on Holter electrocardiography was rapidly increased from 0.84 to 2.24 at the onset of the paroxysmal event, whereas the video-monitoring electroencephalography (EEG) never identified ictal patterns of epileptiform discharges during the episodes. Thus, the diagnosis of PSH was given at 7 years of age. Myoclonic and generalized tonic-clonic seizures frequently appeared from 10 years of age, which poorly responded to anticonvulsants. EEG showed diffuse slow-wave bursts with multifocal spikes. Serial head magnetic resonance imaging (MRI) revealed diffuse cerebral and hippocampal atrophy, but not inflammatory lesions in the limbic system.

Conclusion

We first demonstrate a pediatric case with PSH who developed drug-resistant epilepsy 3 years after the onset of PSH. Our data suggest the pathophysiological link of persistent PSH with chemotherapy-associated brain damage.



中文翻译:

化疗相关脑损伤中的阵发性交感神经过度活跃和癫痫的后期发展

背景

儿童白血病的化疗可能会诱发脑损伤和神经系统后遗症。阵发性交感神经过度活跃 (PSH) 被称为治疗相关并发症。然而,PSH 的完整临床谱仍然难以捉摸。

案例报告

一名 5 岁女孩被诊断出患有急性髓性白血病 (AML) M5。在强化治疗后,她出现了持续数小时的阵发性心动过速、高血压和出汗的反复症状。在发作性事件开始时,动态心电图的低频-高频比从 0.84 迅速增加到 2.24,而视频监测脑电图 (EEG) 从未发现发作期间癫痫样放电的发作模式。因此,PSH 的诊断是在 7 岁时给出的。肌阵挛和全身性强直-阵挛性癫痫发作通常从 10 岁开始出现,对抗惊厥药反应不佳。脑电图显示具有多灶性尖峰的弥漫性慢波爆发。连续头颅磁共振成像 (MRI) 显示弥漫性脑和海马萎缩,

结论

我们首先展示了一个患有 PSH 的儿科病例,该病例在 PSH 发病 3 年后发展为耐药性癫痫。我们的数据表明持续性 PSH 与化疗相关脑损伤的病理生理联系。

更新日期:2021-07-21
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