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β珠蛋白基因突变对印度β地中海贫血临床表型的影响。

Impact of beta globin gene mutations on the clinical phenotype of beta thalassemia in India.

作者信息

Colah Roshan, Nadkarni Anita, Gorakshakar Ajit, Phanasgaonkar Supriya, Surve Reema, Subramaniam P G, Bondge Nagnath, Pujari Kamala, Ghosh Kanjaksha, Mohanty Dipika

机构信息

Institute of Immunohaematology (ICMR), K.E.M. Hospital Campus, Parel, Mumbai 400012, India.

出版信息

Blood Cells Mol Dis. 2004 Sep-Oct;33(2):153-7. doi: 10.1016/j.bcmd.2004.05.002.

Abstract

The beta thalassemias are one of the commonest group of autosomal recessive disorders in India. Although majority of patients are severe and transfusion-dependent, about 10-15% of cases have a milder phenotype. We evaluated the role of beta gene mutations in modulating the clinical presentation of 342 beta thalassemia patients which included 278 severe thalassemia major (TM) and 64 thalassemia intermedia (TI) cases (severe TI: 27; mild TI: 37) from this region. Thirteen beta thalassemia mutations were characterized by reverse dot blot hybridization or amplification refractory mutation system (ARMS); denaturing gradient gel electrophoresis (DGGE) analysis and DNA sequencing helped to characterize the remaining nine mutations. Majority of the patients in the thalassemia major and thalassemia intermedia groups had severe beta+ or beta0 mutations. IVS 1-5 (G-->C) was the commonest mutation in the three groups. The six severe and common Indian mutations [(IVS 1-5 (G-->C), 619 bp deletion, IVS 1-1 (G-->T), codons 8/9 (+G), codon 15 (G-->A), codons 41/42 (-CTTT)] accounted for 92.0% of molecular lesions in the thalassemia major group, 86.8% in the severe TI group, and 72.9% in the mild TI group. IVS 1-1 (G-->T) and codon 30 (G-->C) were significantly more common in thalassemia intermedia cases. The mild capsite +1 (A-->C) mutation was present in both severe and mild cases. Three other mild beta+ mutations, poly A (T-->C), -28 (A-->G), and -88 (C-->T), were seen only in the thalassemia intermedia cases. These four mild mutations in combination with other severe beta+ or beta0 mutations resulted in a very variable clinical presentation. This study reveals that, in majority of Indian patients, the beta genotype cannot predict the phenotype.

摘要

β地中海贫血是印度最常见的常染色体隐性疾病之一。虽然大多数患者病情严重且依赖输血,但约10% - 15%的病例具有较温和的表型。我们评估了β基因突变在调节342例β地中海贫血患者临床表现中的作用,这些患者包括来自该地区的278例重型地中海贫血(TM)和64例中间型地中海贫血(TI)患者(重型TI:27例;轻型TI:37例)。通过反向点杂交或扩增阻滞突变系统(ARMS)对13种β地中海贫血突变进行了鉴定;变性梯度凝胶电泳(DGGE)分析和DNA测序有助于鉴定其余9种突变。重型地中海贫血组和中间型地中海贫血组的大多数患者都有严重的β⁺或β⁰突变。IVS 1 - 5(G→C)是三组中最常见的突变。六种严重且常见的印度突变[IVS 1 - 5(G→C)、619 bp缺失、IVS 1 - 1(G→T)、密码子8/9(+G)、密码子15(G→A)、密码子41/42(-CTTT)]在重型地中海贫血组的分子病变中占92.0%,在重型TI组中占86.8%,在轻型TI组中占72.9%。IVS 1 - 1(G→T)和密码子30(G→C)在中间型地中海贫血病例中明显更常见。轻度帽位点+1(A→C)突变在重型和轻型病例中均有出现。另外三种轻度β⁺突变,多聚A(T→C)、-28(A→G)和-88(C→T),仅在中间型地中海贫血病例中出现。这四种轻度突变与其他严重的β⁺或β⁰突变相结合,导致了非常多样化的临床表现。这项研究表明,在大多数印度患者中,β基因型无法预测表型。

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