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镰状细胞贫血症的分子诊断和产前咨询:SGPGI 经验。

Sickle cell anemia--molecular diagnosis and prenatal counseling: SGPGI experience.

机构信息

Department of Genetics, Sanjay Gandhi Post Graduate Institute of Medical Sciences, Lucknow, 226014 Uttar Pradesh, India.

出版信息

Indian J Pediatr. 2012 Jan;79(1):68-74. doi: 10.1007/s12098-011-0510-1. Epub 2011 Jun 29.

Abstract

OBJECTIVE

To study the issues and dilemmas in prenatal diagnosis of Sickle cell anemia (SCA) and to evaluate the role of genetic modifiers in counseling the families.

METHODS

The authors studied the genotype in 47 individuals with increased HbS and three representative families were taken as an example for describing various issues which need to be sorted out for appropriate counseling.

RESULTS

Of 47 individuals 24 were S beta thalassemia, 14 were homozygous sickle cell anemia (SS) and 9 were HbS trait. In the S beta thalassemia and homozygous SS cases, anemia was presenting manifestation in all. The transfusion requirement in these varied from 0-12 transfusions/ year. Hepatosplenomegaly was seen in 27 cases (71%) and only splenomegaly in 9 cases (23.7%). Jaundice was observed in 34 cases (84.2%). All the 47 subjects (including HbS trait) were studied by Hb Variant system and underwent DNA analysis for beta globin gene mutations, alpha globin gene number and XmnI polymorphism. One or two alpha gene deletion of 3.7 kb (-α3.7/αα or -α3.7/-α3.7) was found in 11 out of 47 cases whereas alpha triplication was found in 2 cases. 28 cases were heterozygous (+/-) for XmnI polymorphism, 9 were homozygous negative (-/-) and 10 were homozygous positive (+/+). Patients with SCA co-inherited with α-thalassemia have less hemolysis as revealed by lower reticulocyte counts than with normal alpha genotype. The authors further discuss the issues and dilemmas faced during prenatal counseling of three families during this study.

CONCLUSIONS

The knowledge of the relationship between genotype and phenotype, effect of the modifier genes has an important role in genetic counseling and for planning individualized treatment for sickle cell anemia.

摘要

目的

研究产前诊断镰状细胞贫血(SCA)的问题和困境,并评估遗传修饰因子在咨询家庭中的作用。

方法

作者研究了 47 例 HbS 增加的个体的基因型,并以三个有代表性的家系为例,描述了需要澄清的各种问题,以便进行适当的咨询。

结果

在 47 例个体中,24 例为 S 珠蛋白生成障碍性贫血,14 例为纯合子镰状细胞贫血(SS),9 例为 HbS 特征。在 S 珠蛋白生成障碍性贫血和纯合子 SS 病例中,贫血是所有病例的表现。这些病例的输血需求从 0-12 次/年不等。肝脾肿大见于 27 例(71%),仅脾肿大见于 9 例(23.7%)。黄疸见于 34 例(84.2%)。所有 47 例(包括 HbS 特征)均通过 Hb 变体系统进行研究,并进行β珠蛋白基因突变、α珠蛋白基因数量和 XmnI 多态性的 DNA 分析。在 47 例病例中有 11 例发现一个或两个 3.7kb 的α基因缺失(-α3.7/αα 或 -α3.7/-α3.7),2 例发现α三倍体。28 例为 XmnI 多态性杂合子(+/-),9 例为纯合子阴性(-/-),10 例为纯合子阳性(+/+)。SCA 合并α-地中海贫血的患者,由于网织红细胞计数较低,溶血程度低于正常α基因型。作者进一步讨论了在本研究中对三个家系进行产前咨询时所面临的问题和困境。

结论

基因型与表型之间的关系以及修饰基因的作用的知识,在遗传咨询和规划镰状细胞贫血的个体化治疗中具有重要作用。

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