Harteveld C L, Losekoot M, Haak H, Heister G A, Giordano P C, Bernini L F
Department of Human Genetics, Leiden University, The Netherlands.
Br J Haematol. 1994 May;87(1):139-43. doi: 10.1111/j.1365-2141.1994.tb04883.x.
In a family of Indian origin we have identified a deletion of two bases at the polyadenylation signal sequence of the alpha 2-globin gene (AATAAA-->AATA). Three individuals heterozygous for this mutation display an alpha o-thalassaemia-like phenotype. Single-stranded conformation analysis and automatic sequencing showed no additional mutations in either alpha 1- or alpha 2-globin genes. A previously described polyadenylation sequence mutation (AATAAA-->AATAAG), alpha TSaudi alpha, causes HbH disease in homozygotes. In this study the patients heterozygous for the AATA(-AA) mutation show a similar phenotype observed in the alpha TSaudi alpha heterozygotes. This confirms the observation that the inefficient transcriptional termination due to mutations of the polyadenylation sequence of the alpha 2-gene might interfere with the alpha 1-gene expression.
在一个印度裔家族中,我们发现α2-珠蛋白基因的聚腺苷酸化信号序列存在两个碱基的缺失(AATAAA→AATA)。三名该突变的杂合子个体表现出αo-地中海贫血样表型。单链构象分析和自动测序显示α1-或α2-珠蛋白基因中无其他突变。先前描述的聚腺苷酸化序列突变(AATAAA→AATAAG),即αTSaudiα,在纯合子中会导致血红蛋白H病。在本研究中,AATA(-AA)突变的杂合子患者表现出与αTSaudiα杂合子相似的表型。这证实了以下观察结果:由于α2-基因聚腺苷酸化序列的突变导致的转录终止效率低下可能会干扰α1-基因的表达。