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与转钴胺素 I(触珠蛋白)的轻度和重度缺乏相关的基因组突变,导致血清钴胺素水平轻度和重度降低。

Genomic mutations associated with mild and severe deficiencies of transcobalamin I (haptocorrin) that cause mildly and severely low serum cobalamin levels.

机构信息

Department of Medicine, New York Methodist Hospital, Brooklyn, NY 11215, USA.

出版信息

Br J Haematol. 2009 Nov;147(3):386-91. doi: 10.1111/j.1365-2141.2009.07855.x. Epub 2009 Aug 17.

Abstract

Transcobalamin (TC) I deficiency, like the function of TC I itself, is incompletely understood. It produces low serum cobalamin levels indistinguishable from those of true cobalamin deficiency. Diagnosis is especially elusive when TC I deficiency is mild. To provide new, more substantive definition, the TCN1 gene was examined in two well-characterised families that included members with both severe and mild TC I deficiencies. A severely deficient proposita with undetectable TC I levels displayed compound heterozygosity for two mutations, each causing a premature stop codon. Relatives in both families who had mildly low or low-normal plasma levels of TC I and cobalamin were heterozygous for one or the other of these mutations. An unrelated patient with mild TC I deficiency and unknown familial TC I and cobalamin status was then tested and found to be similarly heterozygous for one of the mutations. The two nonprivate mutations identify a genetic basis for TC I deficiency for the first time. They also add new approaches to studying mild and severe TC I deficiency and to reducing confusion of its low cobalamin levels with those of cobalamin deficiency and its often dramatically different prognosis and management.

摘要

转钴胺素 (TC) I 缺乏症,就像 TC I 本身的功能一样,尚未被完全理解。它会导致血清钴胺素水平降低,与真正的钴胺素缺乏症难以区分。当 TC I 缺乏症较轻时,诊断尤其难以捉摸。为了提供新的、更实质性的定义,研究人员检查了两个特征明确的家族中的 TCN1 基因,这些家族包括严重和轻度 TC I 缺乏症的成员。一名严重缺乏 TC I 的先证者的 TC I 水平检测不到,显示出两种突变的复合杂合性,每种突变都会导致提前终止密码子。两个家族中 TC I 和钴胺素水平轻度降低或正常偏低的亲属均为这两种突变之一的杂合子。然后对一名 TC I 缺乏症较轻且家族 TC I 和钴胺素状况未知的无关患者进行了检测,发现其也同样为其中一种突变的杂合子。这两种非私有突变首次确定了 TC I 缺乏症的遗传基础。它们还为研究轻度和重度 TC I 缺乏症提供了新的方法,并减少了将其低钴胺素水平与钴胺素缺乏症及其通常截然不同的预后和管理相混淆的情况。

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