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患有脑叶酸缺乏综合征儿童的进行性脑病。

Progressive encephalopathy in a child with cerebral folate deficiency syndrome.

作者信息

Bonkowsky Joshua L, Ramaekers Vincent T, Quadros Edward V, Lloyd Michael

机构信息

Department of Pediatrics, Division of Pediatric Neurology, University of Utah School of Medicine, Salt Lake City, Utah 84108, USA.

出版信息

J Child Neurol. 2008 Dec;23(12):1460-3. doi: 10.1177/0883073808318546. Epub 2008 Oct 14.

Abstract

Cerebral folate deficiency syndrome, a recently recognized cause of developmental delay, regression, and seizures, is associated with autoantibodies against folate receptors. A female child with developmental delay and a history of seizures who presented with seizures and unexplained coma is reported. Extensive testing to evaluate the patient's coma and subsequent developmental regression were unrevealing until the results of her cerebrospinal fluid neurotransmitter analysis returned. These showed low levels of methyltetrahydrofolate, the active metabolite of folate in the cerebrospinal fluid; subsequently, elevated titers of autoantibodies against folate receptors were found. Despite treatment with folinic acid, she developed intractable epilepsy and severe developmental delay.

摘要

脑叶酸缺乏综合征是发育迟缓、倒退和癫痫发作的一种新近认识到的病因,与抗叶酸受体自身抗体有关。本文报告了一名患有发育迟缓且有癫痫发作史的女童,她出现癫痫发作并伴有不明原因的昏迷。在脑脊液神经递质分析结果出来之前,为评估该患者的昏迷及随后的发育倒退所做的广泛检查均未发现异常。这些结果显示脑脊液中叶酸的活性代谢产物甲基四氢叶酸水平较低;随后,发现抗叶酸受体自身抗体滴度升高。尽管接受了亚叶酸治疗,但她仍发展为难治性癫痫和严重发育迟缓。

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N Engl J Med. 2005 May 12;352(19):1985-91. doi: 10.1056/NEJMoa043160.
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Mol Genet Metab. 2005 Apr;84(4):371-3. doi: 10.1016/j.ymgme.2004.12.001. Epub 2005 Jan 22.
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Dev Med Child Neurol. 2004 Dec;46(12):843-51. doi: 10.1017/s0012162204001471.
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