Fujii H, Miwa S
Department of Blood Transfusion Medicine, Tokyo Women's Medical College, Japan.
Am J Hematol. 1990 Aug;34(4):301-10. doi: 10.1002/ajh.2830340412.
During the relatively recent period in which normal genes for most red cell enzymes have been isolated, the techniques of molecular biology have been applied to the studies of erythroenzymopathy. Single nucleotide substitutions have been identified in aldolase, triosephosphate isomerase, glucose 6-phosphate dehydrogenase, and adenylate kinase variants by the cloning and nucleotide sequence of the patients' genes. Up to now, all of the enzyme-deficient variants which have been investigated have been caused by point mutations. An exception is a hemolytic anemia secondary to increased adenosine deaminase (ADA) activity. Red cell ADA activity increases on the order of a hundred-fold in affected individuals. The basic abnormality appears to result from overproduction of structurally normal enzyme due to abnormal transcriptional or translational efficiency.
在大多数红细胞酶的正常基因被分离出来的相对较近的时期,分子生物学技术已被应用于红细胞酶病的研究。通过克隆患者基因并测定核苷酸序列,已在醛缩酶、磷酸丙糖异构酶、葡萄糖6-磷酸脱氢酶和腺苷酸激酶变体中鉴定出单核苷酸替换。到目前为止,所有已研究的酶缺陷变体都是由点突变引起的。一个例外是继发于腺苷脱氨酶(ADA)活性增加的溶血性贫血。在受影响的个体中,红细胞ADA活性增加约100倍。基本异常似乎是由于转录或翻译效率异常导致结构正常的酶过度产生所致。