Successful treatment of infantile epileptic spasms syndrome caused by congenital toxoplasmosis with adrenocorticotropic hormone therapy: A case report

Brain and Development Case Reports Pub Date : 2025-06-11 DOI:10.1016/j.bdcasr.2025.100085

Kentaro Sano , Taku Omata , Yusuke Sasaki , Kenta Ochiai , Megumi Shiota , Shoko Hirose , Yuri Shirato , Naoko Maura , Takashi Kigawa , Ryota Hase , Hisashi Nagase , Masaya Takamoto , Kazumi Norose , Jun-ichi Takanashi

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Abstract

Background

Congenital toxoplasmosis is a rare cause of infantile epileptic spasm syndrome (IESS). Adrenocorticotropic hormone (ACTH) therapy, the first-line treatment for IESS, has the side effect of immunosuppression and may reactivate toxoplasmosis. However, no cases of IESS caused by congenital toxoplasmosis treated with ACTH therapy have been reported.

Case presentation

A 5-month-old Japanese infant presented with epileptic spasms and developmental regression despite partial treatment for toxoplasmosis during fetal life. Brain imaging revealed scattered calcification and polymicrogyria. The serum anti-Toxoplasma gondii IgG level was high. An electroencephalogram showed hypsarrhythmia. ACTH therapy combined with pyrimethamine, sulfadiazine, and folinic acid improved her development and hypsarrhythmia.

Conclusion

IESS caused by congenital toxoplasmosis can be treated without reactivation using ACTH therapy with antiprotozoal drugs.

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促肾上腺皮质激素治疗先天性弓形虫病所致婴儿癫痫性痉挛综合征1例

背景：先天性弓形虫病是引起婴儿癫痫痉挛综合征（IESS）的罕见病因。促肾上腺皮质激素（ACTH）治疗是IESS的一线治疗方法，具有免疫抑制的副作用，并可能重新激活弓形虫病。然而，目前还没有使用ACTH治疗先天性弓形虫病引起的IESS病例的报道。一个5个月大的日本婴儿，尽管在胎儿期对弓形虫病进行了部分治疗，但仍表现出癫痫性痉挛和发育倒退。脑显像显示散在性钙化和多小脑回。血清抗刚地弓形虫IgG水平较高。脑电图显示心律失常。ACTH联合乙胺嘧啶、磺胺嘧啶和亚叶酸改善了她的发育和心律失常。结论采用抗虫药物辅助促肾上腺皮质激素治疗先天性弓形虫病可避免再激活。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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Brain and Development Case Reports

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