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一名15岁女孩的卟啉性脑病:病例报告

Porphyric encephalopathy in a 15-year-old girl: A case report.

作者信息

Dukkipati Saihari S, Terschluse Jordan, Thodeson Drew, Beavers Angela, Muff-Luett Melissa, Ives Claire, Koh Sookyong

机构信息

College of Medicine, University of Nebraska Medical Center, Omaha, NE, USA.

College of Medicine, Creighton University, Omaha, NE, USA.

出版信息

SAGE Open Med Case Rep. 2025 Jan 28;13:2050313X241298532. doi: 10.1177/2050313X241298532. eCollection 2025.

Abstract

A 15-year-old girl presented with new onset tonic-clonic seizures, encephalopathy, abdominal pain, and hypertension with a history of weight loss and emesis. Brain magnetic resonance imaging scans showed diffuse, bilateral cortical and subcortical gray and white matter signal abnormalities. Electroencephalography showed background slowing and disorganization. Extensive evaluation for infection, toxic metabolic, autoimmune disorders, and vasculitis were negative. She was noted to have dark red-colored urine with no red blood cells. Her urine porphobilinogen level was markedly elevated, consistent with the diagnosis of acute intermittent porphyria. She was treated with intravenous hemin with resolution of her neurologic and gastrointestinal symptoms. A hydroxymethylbilane synthase (porphobilinogen deaminase) pathogenic variant was found in porphyria gene panel confirming the diagnosis of acute intermittent porphyria. This case demonstrates a diagnostic challenge posited by a presentation of new onset seizure and encephalopathy due to acute intermittent porphyria, a rare and often overlooked condition in pediatrics.

摘要

一名15岁女孩出现新发强直阵挛性癫痫、脑病、腹痛和高血压,有体重减轻和呕吐史。脑部磁共振成像扫描显示双侧皮质和皮质下灰白质弥漫性信号异常。脑电图显示背景活动减慢和紊乱。对感染、毒性代谢、自身免疫性疾病和血管炎进行的广泛评估均为阴性。她的尿液呈暗红色,但无红细胞。她的尿卟胆原水平显著升高,符合急性间歇性卟啉病的诊断。她接受了静脉注射血红素治疗,神经和胃肠道症状得到缓解。在卟啉病基因检测中发现了一种羟甲基胆色素原合酶(卟胆原脱氨酶)致病变异,确诊为急性间歇性卟啉病。该病例表明,急性间歇性卟啉病导致的新发癫痫和脑病表现带来了诊断挑战,这是儿科一种罕见且常被忽视的疾病。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5ec0/11775956/269a9db1ae2c/10.1177_2050313X241298532-fig1.jpg

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