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

IF 3.3 3区 医学 Q2 TOXICOLOGY Clinical Toxicology Pub Date : 2025-02-01 Epub Date: 2024-12-18 DOI:10.1080/15563650.2024.2418979
Maria Inês de Sá, Filipa Proença
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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.

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一名患有婴儿癫痫痉挛综合征的婴儿在接受维格巴曲林治疗后出现脑磁共振成像异常。
Vigabatrin是一种抗惊厥药物,用于治疗难治性癫痫,并作为婴儿癫痫痉挛综合征的一线治疗药物,很少会引起磁共振成像检测到的大脑异常。这些并发症可能与剂量、年龄和伴随的高剂量促肾上腺皮质激素和/或强的松龙有关,可导致神经系统症状。停药或减量后,症状和影像学改变往往会消失。病例总结:一个7个月大的婴儿被诊断为婴儿癫痫性痉挛综合征,开始服用维加巴特林和强的松龙治疗。然而,痉挛复发,促使维加巴特林剂量的增加和促肾上腺皮质激素的增加,从而减少痉挛的频率。病人后来发展为脑病和上肢震颤。图像:磁共振成像显示丘脑、基底节区、脑干和小脑齿状核的对称性高强度病变,伴扩散受限。结论:我们报告了一名婴儿癫痫痉挛综合征的婴儿用维加巴特林治疗后磁共振成像出现异常。目前除了停药或减量外没有其他治疗方法。
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来源期刊
Clinical Toxicology
Clinical Toxicology 医学-毒理学
CiteScore
5.70
自引率
12.10%
发文量
148
审稿时长
4-8 weeks
期刊介绍: clinical Toxicology publishes peer-reviewed scientific research and clinical advances in clinical toxicology. The journal reflects the professional concerns and best scientific judgment of its sponsors, the American Academy of Clinical Toxicology, the European Association of Poisons Centres and Clinical Toxicologists, the American Association of Poison Control Centers and the Asia Pacific Association of Medical Toxicology and, as such, is the leading international journal in the specialty.
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