Yang K G, Kutlar F, George E, Wilson J B, Kutlar A, Stoming T A, Gonzalez-Redondo J M, Huisman T H
Department of Cell and Molecular Biology, Medical College of Georgia, Augusta 30912-2100.
Br J Haematol. 1989 May;72(1):73-80. doi: 10.1111/j.1365-2141.1989.tb07655.x.
This study concerned the identification of the beta-thalassaemia mutations that were present in 27 Malay patients with Hb E-beta-thalassaemia and seven Malay patients with thalassaemia major who were from West Malaysia. Nearly 50% of all beta-thalassaemia chromosomes carried the G----C substitution at nucleotide 5 of IVS-I; the commonly occurring Chinese anomalies such as the frameshift at codons 41 and 42, the nonsense mutation A----T at codon 17, the A----G substitution at position -28 of the promoter region, and the C----T substitution at position 654 of the second intron, were rare or absent. Two new thalassaemia mutations were discovered. The first involves a frameshift at codon 35 (-C) that was found in two patients with Hb E-beta zero-thalassaemia and causes a beta zero-thalassaemia because a stop codon is present at codon 60. The second is an AAC----AGC mutation in codon 19 that was present on six chromosomes. This substitution results in the production of an abnormal beta chain (beta-Malay) that has an Asn----Ser substitution at position beta 19. Hb Malay is a 'Hb Knossos-like' beta +-thalassaemia abnormality; the A----G mutation at codon 19 likely creates an alternate splicing site between codons 17 and 18, reducing the efficiency of the normal donor splice site at IVS-I to about 60%.
本研究旨在鉴定来自马来西亚西部的27例Hb E-β地中海贫血的马来患者和7例重型地中海贫血的马来患者中存在的β地中海贫血突变。几乎所有β地中海贫血染色体的近50%在IVS-1的第5个核苷酸处携带G→C替换;常见的中国人群中的异常情况,如密码子41和42处的移码突变、密码子17处的无义突变A→T、启动子区域-28位的A→G替换以及第二个内含子654位的C→T替换,在这些患者中很少见或不存在。发现了两种新的地中海贫血突变。第一种涉及密码子35(-C)处的移码突变,在2例Hb E-β0地中海贫血患者中发现,由于密码子60处存在终止密码子,导致β0地中海贫血。第二种是密码子19处的AAC→AGC突变,存在于6条染色体上。这种替换导致产生一种异常的β链(β-马来型),在β19位有Asn→Ser替换。Hb马来型是一种“类Hb Knossos”的β+-地中海贫血异常;密码子19处的A→G突变可能在密码子17和18之间产生一个替代剪接位点,将IVS-1处正常供体剪接位点的效率降低到约60%。