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一名患有婴儿痉挛症综合征的婴儿在接受氨己烯酸治疗后脑部磁共振成像出现异常。

Abnormalities in brain magnetic resonance imaging associated with vigabatrin therapy in an infant with infantile epileptic spasms syndrome.

作者信息

de Sá Maria Inês, Proença Filipa

机构信息

Department of Neuroradiology, Unidade Local de Saúde de Santa Maria, Lisboa, Portugal.

Faculdade de Medicina da Universidade de Lisboa, Clínica Universitária de Imagiologia, Lisboa, Portugal.

出版信息

Clin Toxicol (Phila). 2025 Feb;63(2):137-138. doi: 10.1080/15563650.2024.2418979. Epub 2024 Dec 18.

Abstract

INTRODUCTION

Vigabatrin, an anticonvulsant drug used for refractory epilepsy and as first-line treatment for infantile epileptic spasms syndrome, can rarely cause brain abnormalities detectable on magnetic resonance imaging. These complications, potentially related to dose, young age, and concomitant high doses of adrenocorticotropic hormone and/or prednisolone, can lead to neurological symptoms. Upon withdrawal or dose reduction, symptoms and imaging changes tend to resolve.

CASE SUMMARY

A 7-month-old infant diagnosed with infantile epileptic spasms syndrome started treatment with vigabatrin and prednisolone. However, spasms recurred, prompting an increase in the dose of vigabatrin and the addition of adrenocorticotropic hormone, which reduced the frequency of spasms. The patient later developed encephalopathy and upper limb tremors.

IMAGES

Magnetic resonance imaging revealed symmetrical hyperintense lesions with concomitant restricted diffusion localized in the thalami, basal ganglia, brainstem, and cerebellar dentate nuclei.

CONCLUSION

We report an infant with infantile epileptic spasms syndrome treated with vigabatrin who developed abnormalities on magnetic resonance imaging. There is currently no treatment other than drug withdrawal or reduction.

摘要

引言

氨己烯酸是一种用于治疗难治性癫痫及作为婴儿痉挛症综合征一线治疗药物的抗惊厥药,极少会导致磁共振成像可检测到的脑部异常。这些并发症可能与剂量、年龄小以及同时使用高剂量促肾上腺皮质激素和/或泼尼松龙有关,可导致神经症状。停药或减量后,症状及影像学改变往往会缓解。

病例摘要

一名7个月大被诊断为婴儿痉挛症综合征的婴儿开始使用氨己烯酸和泼尼松龙治疗。然而,痉挛复发,促使增加氨己烯酸剂量并加用促肾上腺皮质激素,这降低了痉挛频率。该患者后来出现脑病和上肢震颤。

影像

磁共振成像显示丘脑、基底神经节、脑干和小脑齿状核出现对称性高信号病变,伴有扩散受限。

结论

我们报告了一名使用氨己烯酸治疗婴儿痉挛症综合征的婴儿出现磁共振成像异常。目前除停药或减量外无其他治疗方法。

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