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II型戊二酸尿症:关于一种此前未被描述的代谢紊乱疾病的报告。

Glutaric aciduria type II: report on a previously undescribed metabolic disorder.

作者信息

Przyrembel H, Wendel U, Becker K, Bremer H J, Bruinvis L, Ketting D, Wadman S K

出版信息

Clin Chim Acta. 1976 Jan 16;66(2):227-39. doi: 10.1016/0009-8981(76)90060-7.

Abstract

A report is given on a hitherto undescribed metabolic disorder, characterized clinically by fatal neonatal acidosis, hypoglycemia and a strong 'sweaty-feet' odour. Biochemical features were a massive urinary excretion of glutaric and lactic acids. Isobutyric, isovaleric and alpha-methylbutyric acids were also greatly increased, followed by adipic, ethylmalonic, alpha-hydroxybutyric, n-butyric, beta-hydroxybutyric, sebacic, suberic, propionic, alpha-hydroxyisovaleric and hexanoic acids. The serum level of glutaric acid was highly elevated. In the serum there were also abnormal levels of lactic, alpha-hydroxybutyric, adipic, suberic, p-hydroxyphenyllactic, myristic, hexadecenoic, palmitic, oleic and stearic acids. Plasma lysine and valine were also elevated. Degradation of 14C-labelled glutaric acid and 14C-labelled branched-chain amino acids, alpha-ketoisovaleric and alpha-ketoisocaproic acids in intact fibroblasts was decreased, whereas that of pyruvic acid was normal. The defect was tentatively supposed to be localized at the level of the metabolism of a range of acyl-CoA compounds. The name glutaric aciduria 'type II' is proposed for the patient's disease.

摘要

报告了一种迄今未被描述的代谢紊乱疾病,其临床特征为致命性新生儿酸中毒、低血糖以及强烈的“汗脚”气味。生化特征为尿中大量排泄戊二酸和乳酸。异丁酸、异戊酸和α-甲基丁酸也显著增加,其次是己二酸、乙基丙二酸、α-羟基丁酸、正丁酸、β-羟基丁酸、癸二酸、辛二酸、丙酸、α-羟基异戊酸和己酸。血清中戊二酸水平高度升高。血清中乳酸、α-羟基丁酸、己二酸、辛二酸、对羟基苯乳酸、肉豆蔻酸、十六碳烯酸、棕榈酸、油酸和硬脂酸水平也异常。血浆中赖氨酸和缬氨酸也升高。完整成纤维细胞中14C标记的戊二酸和14C标记的支链氨基酸、α-酮异戊酸和α-酮异己酸的降解减少,而丙酮酸的降解正常。初步推测该缺陷定位于一系列酰基辅酶A化合物的代谢水平。建议将患者的疾病命名为戊二酸尿症“II型”。

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