Agouti Imane, Badens Catherine, Abouyoub Ahmed, Khattab Mohamed, Sayah Fouad, Barakat Amina, Bennani Mohcine
Laboratoire de Biologie Appliquée, Faculté des Sciences et Techniques, Tanger, Maroc.
Hemoglobin. 2007;31(2):141-9. doi: 10.1080/03630260701285050.
beta-Thalassemia (thal) is the most common recessive inherited disorder in Mediterranean populations. It is estimated that the frequency of this disease in the Moroccan population is between 1.5 and 3.0%. Severe forms of homozygous thalassemia cases require expensive and technically demanding curative (bone marrow transplantation) or palliative (chronic transfusion/chelation) therapies. The -158 (C-->T) polymorphism of the (G)gamma-globin gene (XmnI polymorphism) is known to ameliorate the severity of the disease because of it strong association with an increased production of fetal hemoglobin (Hb F). Among the many known mutations in Morocco, six are common [codon 39 (C-->T), frameshift codon (FSC) 8 (-AA), IVS-II-745 (CG), FSC 6 (-A), -29 (A-->G) and IVS-I-1 (G-->A)]. In this study, we have investigated, in 82 Moroccan beta-thalassemic chromosomes, the correlation between the six common mutations and the XmnI polymorphism using the Fisher exact test. The XmnI polymorphism was divided into two categories, (XmnI [+] and XmnI [-]) and the six common Moroccan mutations into two groups (group I with FSC 8 and group II without FSC 8). Correlation was carried out between the XmnI [+] category and the six common mutations individually that showed that 68% of chromosomes in the XmnI [+] category had the FSC 8 (-AA) mutation. The results reported here show that there is a positive correlation between the XmnI polymorphism and FSC 8 mutation in linkage with haplotype IV [- + - + + - +] (p <10(-5)). In conclusion, molecular determination of genetic markers in early childhood will help to identify candidates for pharmacological Hb F switching by hydroxyurea (HU). In the Moroccan population, a good response to HU treatment should be suspected in cases with the -158 (C-->T) polymorphism in linkage with haplotype IV and internal beta-globin gene framework 3.
β地中海贫血是地中海人群中最常见的隐性遗传性疾病。据估计,摩洛哥人群中这种疾病的发病率在1.5%至3.0%之间。严重的纯合子地中海贫血病例需要昂贵且技术要求高的治愈性(骨髓移植)或姑息性(慢性输血/螯合)治疗。已知(G)γ珠蛋白基因的-158(C→T)多态性(XmnI多态性)可改善疾病的严重程度,因为它与胎儿血红蛋白(Hb F)产量增加密切相关。在摩洛哥众多已知的突变中,有六个是常见的[密码子39(C→T)、移码密码子(FSC)8(-AA)、IVS-II-745(CG)、FSC 6(-A)、-29(A→G)和IVS-I-1(G→A)]。在本研究中,我们使用Fisher精确检验研究了82条摩洛哥β地中海贫血染色体中六个常见突变与XmnI多态性之间的相关性。XmnI多态性分为两类(XmnI [+]和XmnI [-]),六个常见的摩洛哥突变分为两组(第一组有FSC 8,第二组没有FSC 8)。对XmnI [+]类别与六个常见突变分别进行相关性分析,结果显示XmnI [+]类别中68%的染色体具有FSC 8(-AA)突变。此处报告的结果表明,XmnI多态性与FSC 8突变之间存在正相关,与单倍型IV [- + - + + - +]连锁(p <10^(-5))。总之,幼儿期遗传标记的分子测定将有助于识别通过羟基脲(HU)进行药理学Hb F转换的候选者。在摩洛哥人群中,对于与单倍型IV和内部β珠蛋白基因框架3连锁的-158(C→T)多态性病例,应怀疑对HU治疗有良好反应。