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一名5.5个月大患有结节性硬化症所致婴儿痉挛症的女孩出现 vigabatrin 诱发的脑病

Vigabatrin-Induced Encephalopathy in a 5.5-Month-Old Girl with Infantile Spasms due to Tuberous Sclerosis.

作者信息

Klinaki Eleni, Argyri Ioanna, Amountza Georgia, Ioannidou Gerina, Maritsi Despoina, Garoufi Anastasia, Vartzelis George

机构信息

2nd Pediatric Department of National and Kapodistrian University of Athens, Children's Hospital "P. & A. Kyriakou", Athens, Greece.

出版信息

Case Rep Pediatr. 2019 Aug 25;2019:7249237. doi: 10.1155/2019/7249237. eCollection 2019.

Abstract

A 5.5-month-old female infant with tuberous sclerosis complex presented with infantile spasms and was treated with vigabatrin. As her condition did not improve, she was given adrenocorticotropic hormone (ACTH) intramuscularly which stopped the spasms and improved the electroencephalogram (EEG) abnormalities. However, she developed encephalopathy with apathy, drowsiness, and generalized slowing in the EEG. Discontinuation of vigabatrin quickly improved her symptoms and reversed the EEG slowing. A high index of suspicion is required in order to diagnose vigabatrin-induced encephalopathy, especially as the underlying disorders of these patients can be erroneously considered the cause of the observed encephalopathy.

摘要

一名患有结节性硬化症复合体的5.5个月大女婴出现婴儿痉挛,接受了氨己烯酸治疗。由于病情未改善,她接受了肌内注射促肾上腺皮质激素(ACTH),痉挛停止,脑电图(EEG)异常得到改善。然而,她出现了脑病,表现为淡漠、嗜睡,脑电图普遍减慢。停用氨己烯酸后,她的症状迅速改善,脑电图减慢也得到逆转。诊断氨己烯酸诱发的脑病需要高度怀疑,尤其是因为这些患者的基础疾病可能被错误地认为是观察到的脑病的原因。

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