Thalassemia Research Centre, Institute of Molecular Biosciences, Mahidol University, Nakornpathom, Thailand.
Br J Haematol. 2011 Sep;154(5):635-43. doi: 10.1111/j.1365-2141.2011.08770.x. Epub 2011 Jul 7.
Thalassaemia is characterized by the reduced or absent production of globins in the haemoglobin molecule leading to imbalanced α-globin/non α-globin chains. HbE, the result of a G to A mutation in codon 26 of the HBB (β-globin) gene, activates a cryptic 5' splice site in codon 25 leading to a reduction of correctly spliced β(E) -globin (HBB:c.79G>A) mRNA and consequently β(+) -thalassaemia. A wide range of clinical severities in bothα- and β-thalassaemia syndromes, from nearly asymptomatic to transfusion-dependent, has been observed. The correlation between clinical heterogeneity in various genotypes of thalassaemia and the levels of globin gene expression and β(E) -globin pre-mRNA splicing were examined using multiplex quantitative real-time reverse transcription polymerase chain reaction (RT-qPCR) and allele-specific RT-qPCR. The α-globin/non α-globin mRNA ratio was demonstrated to be a good indicator for disease severity among different thalassaemia disorders. However, the α-globin/non α-globin mRNA ratio ranged widely in β-thalassaemia/HbE patients, with no significant difference between mild and severe phenotypes. Interestingly, the correctly to aberrantly spliced β(E) -globin mRNA ratio in 30% of mild β-thalassaemia/HbE patients was higher than that of the severe patients. The splicing process of β(E) -globin pre-mRNA differs among β-thalassaemia/HbE patients and serves as one of the modifying factors for disease severity.
地中海贫血的特征是血红蛋白分子中珠蛋白的产量减少或缺失,导致α-珠蛋白/非α-珠蛋白链不平衡。HbE 是 HBB(β-珠蛋白)基因密码子 26 处的 G 到 A 突变的结果,激活了密码子 25 处的隐蔽 5'剪接位点,导致正确剪接的 β(E)-珠蛋白(HBB:c.79G>A)mRNA 减少,从而导致β(+)地中海贫血。在α-和β-地中海贫血综合征中,观察到从几乎无症状到依赖输血的各种临床严重程度。使用多重实时定量 RT-PCR(RT-qPCR)和等位基因特异性 RT-qPCR 检查了地中海贫血各种基因型的临床异质性与珠蛋白基因表达和β(E)-珠蛋白前体 mRNA 剪接水平之间的相关性。α-珠蛋白/非α-珠蛋白 mRNA 比值被证明是不同地中海贫血疾病严重程度的良好指标。然而,β-地中海贫血/HbE 患者的α-珠蛋白/非α-珠蛋白 mRNA 比值范围很广,轻度和重度表型之间没有显著差异。有趣的是,30%的轻度β-地中海贫血/HbE 患者中正确剪接到异常剪接的β(E)-珠蛋白 mRNA 比值高于重度患者。β(E)-珠蛋白前体 mRNA 的剪接过程在β-地中海贫血/HbE 患者中存在差异,是疾病严重程度的修饰因素之一。