Thong Meow-Keong, Tan J A M A, Tan K L, Yap S F
Department of Paediatrics, Faculty of Medicine, University of Malaya, Kuala Lumpur, Malaysia.
J Trop Pediatr. 2005 Dec;51(6):328-33. doi: 10.1093/tropej/fmi052. Epub 2005 Jun 20.
beta-thalassaemia major, an autosomal recessive hemoglobinopathy, is one of the most common single gene disorders in multi-racial Malaysia. The control of beta-thalassaemia major requires a multi-disciplinary approach that includes population screening, genetic counselling, prenatal diagnosis and the option of termination of affected pregnancies. To achieve this objective, the molecular characterisation of the spectrum of beta-globin gene mutations in each of the affected ethnic groups is required. We studied 88 consecutive unrelated individuals and their respective families with beta-thalassaemia (74 beta-thalassaemia major, 12 HbE-beta-thalassaemia, 2 with HbE homozygotes) and four individuals with beta-thalassaemia trait that contributed a total 180 alleles for study. Using a 2-step molecular diagnostic strategy consisting of amplification refractory mutation system (ARMS) to identify the 8 most common mutations followed by other DNA-based diagnostic techniques, a total of 177 (98.3 per cent) of the 180 beta-thalassaemia alleles were characterised. One out of 91 (1 per cent) of the Chinese alleles, one out of 46 (2.2 per cent) Malay alleles and one out of two Indian alleles remained unknown. A 100 per cent success rate was achieved in studying the Kadazandusun community in this study. A strategy to identify beta-globin gene mutations in Malaysians with beta-thalassaemia is proposed based on this outcome.
重型β地中海贫血是一种常染色体隐性血红蛋白病,是多民族的马来西亚最常见的单基因疾病之一。重型β地中海贫血的控制需要多学科方法,包括人群筛查、遗传咨询、产前诊断以及终止受影响妊娠的选择。为实现这一目标,需要对每个受影响种族群体中β珠蛋白基因突变谱进行分子特征分析。我们研究了88名连续的无关个体及其患有β地中海贫血的各自家庭(74例重型β地中海贫血、12例HbE-β地中海贫血、2例HbE纯合子)以及4例β地中海贫血特征个体,共180个等位基因用于研究。采用两步分子诊断策略,包括使用扩增阻滞突变系统(ARMS)识别8种最常见的突变,随后采用其他基于DNA的诊断技术,180个β地中海贫血等位基因中共有177个(98.3%)得到了特征分析。91个(1%)中国等位基因、46个(2.2%)马来等位基因和2个印度等位基因中的1个仍未明确。在本研究中对卡达山杜顺社区的研究成功率达到了100%。基于这一结果,提出了一种识别马来西亚β地中海贫血患者β珠蛋白基因突变的策略。