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1例MRI表现酷似双侧丘脑梗死的vigabatrin中毒病例。

A Case of Vigabatrin Toxicity Mimicking Bilateral Thalamic Infarcts on MRI.

作者信息

Lockard Trevor J, Rathore Geetanjali

机构信息

Pediatric Neurology, University of Nebraska Medical Center, Omaha, USA.

Pediatric Neurology, Children's Hospital & Medical Center, Omaha, USA.

出版信息

Cureus. 2023 Sep 11;15(9):e45049. doi: 10.7759/cureus.45049. eCollection 2023 Sep.

Abstract

A 20-month-old female with a past medical history of tuberous sclerosis, epilepsy, and infantile spasms treated with vigabatrin presented for surveillance MRI for multiple brain hamartomatous lesions and subependymal nodules. MRI showed new restricted diffusion to bilateral thalami and globi pallidi. This finding was concerning for bilateral thalamic strokes, with differential to include infection, metabolic etiologies, or toxic injuries. Without focal or diffuse neurologic symptoms or additional MRI lesions to suggest an acute or chronic pathology, it was determined the MRI signal changes were likely induced by vigabatrin. Vigabatrin therapy was continued, and a repeat MRI 17 months later showed a resolution of the diffusion restriction with no residual sequelae. Vigabatrin-induced MRI abnormalities are an uncommon adverse effect of therapy for infantile spasms, with adverse events being most common in young infants. It is crucial to consider this adverse drug effect in an asymptomatic patient presenting with these MRI lesions as the findings are otherwise suggestive of a serious disease process, such as an inborn error of metabolism, requiring expensive and invasive workup.

摘要

一名20个月大的女性,有结节性硬化症、癫痫和婴儿痉挛症病史,曾接受vigabatrin治疗,此次因监测多发脑错构瘤病变和室管膜下结节而行MRI检查。MRI显示双侧丘脑和苍白球出现新的扩散受限。这一发现令人担忧双侧丘脑中风,鉴别诊断包括感染、代谢病因或中毒性损伤。由于没有局灶性或弥漫性神经症状,也没有其他MRI病变提示急性或慢性病理情况,因此确定MRI信号改变可能是由vigabatrin引起的。继续使用vigabatrin治疗,17个月后复查MRI显示扩散受限消失,无残留后遗症。Vigabatrin诱导的MRI异常是婴儿痉挛症治疗中一种罕见的不良反应,不良事件在幼儿中最为常见。对于出现这些MRI病变的无症状患者,考虑这种药物不良反应至关重要,因为否则这些发现提示存在严重疾病过程,如先天性代谢缺陷,需要进行昂贵且有创的检查。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/57ce/10566640/6044fd343a13/cureus-0015-00000045049-i01.jpg

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