Goodman S I, Norenberg M D, Shikes R H, Breslich D J, Moe P G
J Pediatr. 1977 May;90(5):746-50. doi: 10.1016/s0022-3476(77)81240-7.
Biochemical and morphologic studies on a patient with glutaric aciduria are presented. Generalized aminoaciduria, alpha-aminoadipic aciduria, and saccharopinuria were noted just prior to death, as well as glutaconic aciduria greater than beta-hydroxyglutaric aciduria. Mutant liver mitochondria did not oxidize glutaryl-CoA to glutaconyl-CoA, indicating deficiency of glytaryl-CoA dehydrogenase. Autopsy revealed cerebral edema, ischemic neuronal changes, and striatal degeneration in the brain with fatty changes in liver, kidney, and myocardium.
本文报告了对一名戊二酸尿症患者的生化和形态学研究。在患者死亡前发现了全身性氨基酸尿、α-氨基己二酸尿和尿苷酸尿,以及戊烯二酸尿大于β-羟基戊二酸尿。突变的肝线粒体不能将戊二酰辅酶A氧化为戊烯二酰辅酶A,表明缺乏戊二酰辅酶A脱氢酶。尸检显示脑水肿、缺血性神经元改变以及大脑纹状体变性,同时肝脏、肾脏和心肌出现脂肪变性。