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腺嘌呤磷酸核糖转移酶缺乏症:一种人类之前未被描述的基因缺陷。

Adenine phosphoribosyltransferase deficiency: a previously undescribed genetic defect in man.

作者信息

Kelley W N, Levy R I, Rosenbloom F M, Henderson J F, Seegmiller J E

出版信息

J Clin Invest. 1968 Oct;47(10):2281-9. doi: 10.1172/JCI105913.

Abstract

A deficiency of adenine phosphoribosyltransferase (A-PRTase) is described in four members in three generations of one family. A-PRTase is coded by an autosome and the mutants described in this report are heterozygotes for this enzyme defect. The level of enzyme activity in these heterozygotes was inappropriately low, ranging from 21 to 37% of normal rather than the expected 50% of normal. Examination of various physical and chemical properties of the A-PRTase obtained from the mutant heterozygotes failed to reveal differences from the normal enzyme. These patients have no discernable abnormality in uric acid production despite the finding that patients with a deficiency of a closely related enzyme, hypoxanthine-guanine phosphoribosyltransferase, invariably produce excessive quantities of uric acid. A relationship of the A-PRTase deficiency to the disturbance in lipoprotein metabolism observed in the propositus has not been firmly established. Possible manifestations of the homozygous form of this enzyme deficiency will require identification of such individuals in the future.

摘要

在一个家族的三代人中,有四名成员被发现缺乏腺嘌呤磷酸核糖转移酶(A-PRTase)。A-PRTase由常染色体编码,本报告中描述的突变体是该酶缺陷的杂合子。这些杂合子中的酶活性水平异常低,仅为正常水平的21%至37%,而非预期的50%。对从突变杂合子中获得的A-PRTase的各种物理和化学性质进行检测,未发现其与正常酶有差异。尽管发现缺乏密切相关的酶——次黄嘌呤-鸟嘌呤磷酸核糖转移酶的患者总是会产生过量尿酸,但这些患者在尿酸生成方面没有明显异常。A-PRTase缺乏与先证者中观察到的脂蛋白代谢紊乱之间的关系尚未得到确凿证实。这种酶缺乏的纯合子形式可能出现的表现,未来需要识别出此类个体才能明确。

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