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葡萄糖-6-磷酸脱氢酶变体:与新生儿黄疸相关的Gd(+) Alexandra以及一名患有板层状白内障的年轻男性中的Gd(-) Camperdown 。

Glucose 6-phosphate dehydrogenase variants: Gd (+) Alexandra associated with neonatal jaundice and Gd (-) Camperdown in a young man with lamellar cataracts.

作者信息

Harley J D, Agar N S, Yoshida A

出版信息

J Lab Clin Med. 1978 Feb;91(2):295-300.

PMID:23402
Abstract

Two male subjects are described, with unusual clinical presentations and with hitherto undescribed G6PD variants. The first, of Italian extraction, suffered from severe neonatal jaundice following maternal ingestion of fresh broad beans (Vicia fava) both prenatally and postnatally: the expression of the enzymatic defect was much more severe in the neonatal period than on retesting in adolescence, when biochemical characterization showed unique features which justify designation as a new variant Gd(+) Alexandra. The second patient, a boy of Maltese extraction who was found to have bilateral lamellar cataracts at the age of 4 years, was identified as G6PD deficient only as a result of a survey of children of Mediterranean origin with unexplained cataract formation; he has approximately 15% of normal enzyme activity, with another unique combination of biochemical characteristics which has led to its designation as Gd(-) Camperdown. Although this association may be coincidental, it prompts further attention to the possibility that under certain circumstances G6PD deficiency may favor cataract formation. The two cases illustrate the value of characterization of the mutant enzyme whenever unexpected clinical or laboratory results are obtained.

摘要

本文描述了两名男性受试者,他们具有不同寻常的临床表现以及迄今未被描述过的葡萄糖-6-磷酸脱氢酶(G6PD)变体。第一名受试者为意大利裔,其母亲在产前和产后食用新鲜蚕豆(蚕豆属)后,该受试者患上了严重的新生儿黄疸:酶缺陷在新生儿期的表现比青春期再次检测时更为严重,青春期生化特征显示出独特特征,这使其被认定为一种新的变体Gd(+) Alexandra。第二名患者是一名马耳他裔男孩,4岁时被发现患有双侧板层白内障,仅在对患有不明原因白内障的地中海裔儿童进行调查时才被确定为G6PD缺乏;他的酶活性约为正常水平的15%,其生化特征的另一种独特组合使其被命名为Gd(-) Camperdown。尽管这种关联可能是巧合,但它促使人们进一步关注在某些情况下G6PD缺乏可能会促进白内障形成的可能性。这两个病例说明了每当获得意外的临床或实验室结果时,对突变酶进行特征分析的价值。

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