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蛋白质摄入量急性增加引发的迟发性鸟氨酸转氨甲酰酶缺乏症:文献报道的10例病例综述

Late Onset Ornithine Transcarbamylase Deficiency Triggered by an Acute Increase in Protein Intake: A Review of 10 Cases Reported in the Literature.

作者信息

Barkovich E, Gropman A L

机构信息

Department of Radiology, George Washington University School of Medicine and Health Sciences, Washington, DC, USA.

Department of Neurology, George Washington University School of Medicine and Health Sciences, Children's National Medical Center, Washington, DC, USA.

出版信息

Case Rep Genet. 2020 Apr 25;2020:7024735. doi: 10.1155/2020/7024735. eCollection 2020.

DOI:10.1155/2020/7024735
PMID:32373372
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7197010/
Abstract

While the urea cycle disorders (UCDs) classically present in the neonatal stage, they have become increasingly recognized as a rare cause of unexplained hyperammonemic encephalopathy in adults. Many metabolic triggers for late-onset UCDs have been described in the literature including excessive protein intake. In this case series, ten such documented cases are reviewed with analysis of patient demographic, protein load, treatment course, and patient outcome. Common delays in treatment include recognition of hyperammonemia as the cause of encephalopathy and initiation of hemodialysis. In only one case was a diet history used to raise suspicion for a metabolic derangement. Metabolic disorders remain an important consideration in adults presenting with encephalopathy not explained by more common etiologies, and recent and remote dietary history may provide valuable information.

摘要

虽然尿素循环障碍(UCDs)典型地在新生儿期出现,但它们已越来越被认为是成人不明原因高氨血症性脑病的罕见病因。文献中已描述了许多迟发性UCDs的代谢触发因素,包括蛋白质摄入过多。在本病例系列中,回顾了10例此类有记录的病例,并分析了患者的人口统计学特征、蛋白质负荷、治疗过程和患者结局。常见的治疗延迟包括将高氨血症识别为脑病的病因以及开始血液透析。仅在1例病例中,饮食史被用于引发对代谢紊乱的怀疑。代谢紊乱仍然是患有无法用更常见病因解释的脑病的成人的重要考虑因素,近期和既往饮食史可能提供有价值的信息。

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本文引用的文献

1
Late-onset ornithine transcarbamylase deficiency: An under recognized cause of metabolic encephalopathy.迟发性鸟氨酸转氨甲酰酶缺乏症:一种未被充分认识的代谢性脑病病因。
SAGE Open Med Case Rep. 2014 Jul 31;2:2050313X14546348. doi: 10.1177/2050313X14546348. eCollection 2014.
2
Fatal coma in a young adult due to late-onset urea cycle deficiency presenting with a prolonged seizure: a case report.一例迟发性尿素循环障碍导致青年成人出现长时间癫痫发作并致死性昏迷的病例报告
J Med Case Rep. 2015 Nov 23;9:267. doi: 10.1186/s13256-015-0741-2.
3
Distinct phenotype of hepatotoxicity associated with illicit use of anabolic androgenic steroids.与非法使用合成代谢雄激素类固醇相关的肝毒性的独特表型。
Aliment Pharmacol Ther. 2015 Jan;41(1):116-25. doi: 10.1111/apt.13023. Epub 2014 Nov 13.
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Suggested guidelines for the diagnosis and management of urea cycle disorders.尿素循环障碍的诊断和管理建议指南。
Orphanet J Rare Dis. 2012 May 29;7:32. doi: 10.1186/1750-1172-7-32.
5
Hyperammonemia in a patient with late-onset ornithine carbamoyltransferase deficiency.患者患有迟发性鸟氨酸氨甲酰基转移酶缺陷症,伴有高血氨症。
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Fatal ammonia toxicity in an adult due to an undiagnosed urea cycle defect: under-recognition of ornithine transcarbamylase deficiency.成人因未诊断的尿素循环缺陷导致致命性氨毒性:精氨酸代琥珀酸合成酶缺乏症认识不足。
Ann Clin Biochem. 2010 May;47(Pt 3):279-81. doi: 10.1258/acb.2010.009250. Epub 2010 Apr 20.
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Acute hyperammonemic encephalopathy in adult onset ornithine transcarbamylase deficiency.成人起病型鸟氨酸转氨甲酰酶缺乏症所致急性高氨血症性脑病
Intensive Care Med. 2008 Oct;34(10):1922-4. doi: 10.1007/s00134-008-1217-2. Epub 2008 Jul 24.
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