Barkovich E, Gropman A L
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.
Case Rep Genet. 2020 Apr 25;2020:7024735. doi: 10.1155/2020/7024735. eCollection 2020.
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.
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。