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人类葡萄糖-6-磷酸转运酶基因的基因组结构及一名日本糖原贮积病Ib型患者该基因的新突变。

Genomic structure of the human glucose 6-phosphate translocase gene and novel mutations in the gene of a Japanese patient with glycogen storage disease type Ib.

作者信息

Ihara K, Kuromaru R, Hara T

机构信息

Department of Pediatrics, Faculty of Medicine, Kyushu University, Fukuoka, Japan.

出版信息

Hum Genet. 1998 Oct;103(4):493-6. doi: 10.1007/s004390050856.

Abstract

Glycogen storage disease (GSD) type Ib is an autosomal recessive disorder caused by a deficiency in microsomal glucose 6-phosphate (G6P) translocase. A gene mutated in GSD type Ib patients has recently been isolated. We have determined the entire sequence of the human G6P translocase gene by PCR-directed sequencing. The gene spans approximately 5 kb of genomic DNA and contains eight exons. Analysis of DNA from a Japanese patient with GSD type Ib revealed new compound heterozygous mutations; a T to C transition at cDNA position 521 resulting in W118R, and an A to C transversion at the -2 splicing acceptor site of intron 1. Reverse transcription (RT)-PCR from leukocyte RNA of the patient revealed the abnormally spliced transcript. These results further support the suggestion that the gene is causative for GSD Ib and should be useful in the molecular diagnosis of such patients.

摘要

Ⅰb型糖原贮积病(GSD)是一种常染色体隐性疾病,由微粒体葡萄糖6 - 磷酸(G6P)转位酶缺乏引起。最近已分离出在Ⅰb型糖原贮积病患者中发生突变的基因。我们通过PCR定向测序确定了人类G6P转位酶基因的完整序列。该基因跨越约5kb的基因组DNA,包含8个外显子。对一名日本Ⅰb型糖原贮积病患者的DNA分析发现了新的复合杂合突变;cDNA位置521处的T到C转换导致W118R,以及内含子1的 - 2剪接受体位点处的A到C颠换。从患者白细胞RNA进行的逆转录(RT)-PCR显示了异常剪接的转录本。这些结果进一步支持了该基因是Ⅰb型糖原贮积病病因的观点,并且应该有助于对此类患者进行分子诊断。

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