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西班牙首例磷酸果糖激酶缺乏症的描述:PFKM 基因中一种新型纯合错义突变的鉴定。

First description of phosphofructokinase deficiency in spain: identification of a novel homozygous missense mutation in the PFKM gene.

机构信息

Red Cell Pathology Unit, Biomedical Dianostic Centre, University Hospital Clínic de Barcelona Barcelona, Spain.

Faculty of Medicine and Dentistry, Department of Biology, Palacky University Olomouc, Czech Republic.

出版信息

Front Physiol. 2013 Dec 30;4:393. doi: 10.3389/fphys.2013.00393. eCollection 2013.

Abstract

Phosphofructokinase deficiency is a very rare autosomal recessive disorder, which belongs to group of rare inborn errors of metabolism called glycogen storage disease. Here we report on a new mutation in the phosphofructokinase (PFK) gene PFKM identified in a 65-years-old woman who suffered from lifelong intermittent muscle weakness and painful spasms of random occurrence, episodic dark urines, and slight haemolytic anemia. After ruling out the most common causes of chronic haemolytic anemia, the study of a panel of 24 enzyme activities showed a markedly decreased PFK activity in red blood cells (RBCs) from the patient. DNA sequence analysis of the PFKM gene subsequently revealed a novel homozygous mutation: c.926A>G; p.Asp309Gly. This mutation is predicted to severely affect enzyme catalysis thereby accounting for the observed enzyme deficiency. This case represents a prime example of classical PFK deficiency and is the first reported case of this very rare red blood cell disorder in Spain.

摘要

磷酸果糖激酶缺乏症是一种非常罕见的常染色体隐性遗传病,属于一类称为糖原贮积病的罕见先天性代谢错误。在这里,我们报告了在一位 65 岁女性中发现的磷酸果糖激酶(PFK)基因 PFKM 的一个新突变,该女性患有终生间歇性肌肉无力和随机发生的疼痛性痉挛、间歇性深色尿液和轻微溶血性贫血。在排除了慢性溶血性贫血的最常见原因后,对 24 种酶活性的研究表明,患者的红细胞(RBC)中 PFK 活性明显降低。随后对 PFKM 基因的 DNA 序列分析显示了一种新的纯合突变:c.926A>G;p.Asp309Gly。该突变预计会严重影响酶的催化作用,从而导致观察到的酶缺乏。该病例代表了经典 PFK 缺乏症的一个典型例子,也是西班牙首例该非常罕见的红细胞疾病的报道。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b7a2/3875906/27e0f2e9823c/fphys-04-00393-g0001.jpg

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