Acute encephalopathy with biphasic seizures and late reduced diffusion accompanied by Takotsubo cardiomyopathy
Acute encephalopathy with biphasic seizures and late reduced diffusion accompanied by Takotsubo cardiomyopathy
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DOI：
10.1016/j.braindev.2018.10.002
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发表时间：
2019-03-01
影响因子：
1.7
通讯作者：
Iijima, Kazumoto
中科院分区：
文献类型：
--
作者：
Yamaguchi, Hiroshi;Nagase, Hiroaki;Iijima, Kazumoto
工作背景：急性脑病伴双相癫痫发作和迟发性弥散减低（AESD）的特征是双相癫痫发作和意识障碍。Takotsubo心肌病（TTC），这是典型的触发心理或身体的压力，其特点是短暂的心肌功能障碍，影响左心室心尖。最近的报告表明，癫痫发作也可能引发TTC。然而，没有TTC的情况下，伴随着AESD.Patient：一个以前健康的4岁女孩被带到医院与第一次发热全身强直阵挛性惊厥，持续约40分钟。癫痫发作后，她被插管，由于呼吸恶化。第二天，她的心脏功能恶化，超声心动图显示左心室收缩期心尖气球样变伴基底壁运动过度，这是TTC患者的典型特征。她的病情逐渐好转，并逐渐减少儿茶酚胺支持。然而，入院后6天，她出现了一系列短暂的抽搐。入院后10天，头部MRI显示除枕叶外，整个皮质扩散减少的病变，以及周围保留。发病后35天的随访MRI显示全脑萎缩，随后她开发了严重的认知功能障碍。结论：我们的病人发展TTC伴随AESD的功能。因此，我们的研究结果可能会提供深入了解TTC的发展，并促进进一步研究癫痫发作和TTC之间的关系。(C)2018日本儿童神经病学学会Elsevier B.V.出版，保留所有权利。
Background: Acute encephalopathy with biphasic seizures and late reduced diffusion (AESD) is characterized by biphasic seizures and impaired consciousness. Takotsubo cardiomyopathy (TTC), which is typically triggered by psychological or physical stress, is characterized by transient myocardial dysfunction affecting the left ventricular apex. Recent reports have suggested that seizures can also trigger TTC. However, no cases of TTC accompanied by AESD have been reported.Patient: A previously healthy 4-year-old girl was brought to a hospital with first-time febrile generalized tonic-clonic convulsions, which lasted approximately 40 min. After the seizure resolved, she was intubated due to respiratory deterioration. On the next day, her cardiac function deteriorated, and echocardiography revealed systolic apical ballooning of the left ventricle accompanied by hyperkinesis of the basal wall, which are typical in patients with TTC. Her condition gradually improved, and catecholamine support was tapered. However, 6 days after admission, she experienced a cluster of brief convulsions. Ten days after admission, head MRI revealed lesions with reduced diffusion throughout the cortex, except in the occipital lobe, as well as perirolandic sparing. Follow-up MRI 35 days after onset revealed whole-brain atrophy, following which she developed severe cognitive dysfunction.Conclusions: Our patient developed TTC accompanied by features of AESD. Our findings may thus provide insight into the development of TTC and prompt further studies regarding the relationship between prolonged seizures and TTC. (C) 2018 The Japanese Society of Child Neurology. Published by Elsevier B.V. All rights reserved.