Adekile A D, Kutlar F, Haider M Z, Kutlar A
Department of Pediatrics, Kuwait University, Kuwait.
Am J Hematol. 2001 Apr;66(4):263-6. doi: 10.1002/ajh.1055.
Sickle cell disease (SCD) is relatively mild among Kuwaiti Arabs. However, an atypical subset of patients exists with frequent, severe vaso-occlusive crisis and osteonecrosis. The thermolabile variant of MTHFR, resulting from a C-->T mutation at nucleotide 677, has been shown to be associated with hyperhomocysteinemia, which is an important risk factor for premature vascular disease. We have screened an unselected group of 41 Kuwaiti SCD patients (33 SS and 8 Sbeta(0)-thal) attending the Hematology Clinic of Kuwait University Teaching Hospital for the MTHFR mutation, using a PCR-RFLP method. The patients were aged 2-41 years (mean of 12.8 +/- 8.6). One (2.4%) individual was homozygous for the mutation while 15 (36.6%) were heterozygous, giving an allele frequency of 20.7%. Twenty-one patients (14 SS and 7 Sbeta(0)-thal) were screened for osteonecrosis using MRI of the hip (spin-echo T1- and T2-weighted images). Seven (33.3%) had varying degrees of osteonecrosis, among whom the frequency of the 677 C-->T allele was 21.4%. The frequency was identical among those without osteonecrosis. Although the allele frequency is higher among our patients compared to American SS patients, our results do not suggest an association with osteonecrosis.
镰状细胞病(SCD)在科威特阿拉伯人中相对较轻。然而,存在一个非典型的患者亚群,他们频繁发生严重的血管闭塞性危机和骨坏死。亚甲基四氢叶酸还原酶(MTHFR)的热不稳定变体是由核苷酸677处的C→T突变产生的,已被证明与高同型半胱氨酸血症有关,高同型半胱氨酸血症是过早发生血管疾病的一个重要危险因素。我们使用聚合酶链反应-限制性片段长度多态性(PCR-RFLP)方法,对科威特大学教学医院血液科门诊的41例未经选择的科威特SCD患者(33例SS型和8例Sβ0-地中海贫血型)进行了MTHFR突变筛查。患者年龄在2至41岁之间(平均12.8±8.6岁)。1例(2.4%)个体为该突变的纯合子,15例(36.6%)为杂合子,等位基因频率为20.7%。对21例患者(14例SS型和7例Sβ0-地中海贫血型)进行了髋关节磁共振成像(自旋回波T1加权和T2加权图像)筛查骨坏死。7例(33.3%)有不同程度的骨坏死,其中677 C→T等位基因频率为21.4%。在无骨坏死的患者中该频率相同。虽然我们患者中的等位基因频率高于美国的SS型患者,但我们的结果并不表明与骨坏死有关。