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鸟氨酸氨甲酰基转移酶缺乏症中的嘧啶和嘌呤代谢物。

Pyrimidine and purine metabolites in ornithine carbamoyl transferase deficiency.

作者信息

Webster D R, Simmonds H A, Barry D M, Becroft D M

出版信息

J Inherit Metab Dis. 1981;4(1):27-31. doi: 10.1007/BF02263578.

Abstract

Detailed biochemical studies have been carried out in a female heterozygote for ornithine carbamoyl-transferase (OCT) deficiency. Increased levels of the pyrimidines, orotic acid, uridine and uracil, were observed in plasma as well as urine by utilizing an adaptation of high performance liquid chromatography (HPLC). Urinary clearances of these compounds were high, that of orotic acid indicating net secretion. Urinary uric acid clearance was also elevated, a finding attributed to the uricosuric effect of the orotic acid excreted concomitantly. The results in this child and her family are typical of OCT deficiency. They confirm considerable genetic heterogeneity in the biochemical as well as clinical expression in this defect.

摘要

对一名鸟氨酸氨甲酰基转移酶(OCT)缺乏症的女性杂合子进行了详细的生化研究。通过采用高效液相色谱法(HPLC)的改良方法,在血浆和尿液中均观察到嘧啶、乳清酸、尿苷和尿嘧啶水平升高。这些化合物的尿清除率很高,乳清酸的尿清除率表明有净分泌。尿尿酸清除率也升高,这一发现归因于同时排出的乳清酸的促尿酸尿作用。该患儿及其家族的结果是OCT缺乏症的典型表现。它们证实了该缺陷在生化及临床表型上存在相当大的遗传异质性。

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