广西壮族自治区遗传性球形红细胞增多症的基因突变分析。

IF 0.6 4区 医学 Q4 HEMATOLOGY
Journal of Hematopathology Pub Date : 2023-06-01 Epub Date: 2023-05-05 DOI:10.1007/s12308-023-00545-8
Xingyuan Chen, Lin Liao, Yangyang Wu, Liqun Xiang, Yumei Qin, Meiling Luo, Faquan Lin
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引用次数: 0

摘要

遗传性球形红细胞增多症(HS)是一种常见的遗传性溶血性贫血(HHA),是由五种红细胞膜蛋白紊乱引起的。HS 在中国广西也很常见。研究人员利用靶区捕获高通量测序技术分析了在HS患者中发现的基因突变。在某些病例中还进行了谱系分析,为复杂遗传性溶血性贫血的病因诊断提供了优化方法。我们通过实验室检测、靶区捕获高通量测序技术和桑格测序技术对患者及其家属的血样进行了评估。我们从 37 个无血缘关系的家庭中发现了 79 名 HS 患者。在这些患者身上观察到的突变主要存在于四个与 HS 相关的基因中。这些基因包括:SLC4A1(31.65%的患者(25/79)发生突变)、SPTA1(30.78%(24/79))、EPB42(6.33%(5/79))和SPTB(5.06%(4/79))。26.58%(21/79)的患者出现复合基因型,包括两个或多个 HS 相关基因突变,或 HS 相关基因突变合并地中海贫血或 G6PD 缺乏。除总胆红素外,不同基因型患者的临床症状无明显差异。复合基因型的平均网织红细胞体积(MRV)和平均球形细胞体积(MSCV)与其他组别有显著差异。在 HS 相关基因中共发现 28 种突变类型。利用高通量测序技术,我们还发现了一些被误诊的病例。MRV和MSCV作为HS的敏感决定因素,在复合突变中更为重要。高通量测序技术可为HS提供更有效的病因诊断方法,具有高效性和特异性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Genetic mutation analysis of hereditary spherocytosis in Guangxi Zhuang Autonomous Region.

Hereditary spherocytosis (HS) is a common, hereditary hemolytic anemia (HHA) that is attributed to the disturbance of five erythrocyte membrane proteins. HS is also common in Guangxi, China. Target region capture high-throughput sequencing technology was used to analyze genetic mutations found in HS patients. Pedigree analysis was also performed, in some cases, to provide an optimized approach for the etiological diagnosis of complex, hereditary hemolytic anemia. Blood samples from the probands and their families were assessed by laboratory tests, target region capture high-throughput sequencing technology, and Sanger sequencing. We detected 79 HS patients from 37 unrelated families. The mutations observed in these patients were found mainly in four HS-related genes. These included SLC4A1, which was mutated in 31.65% of patients (25/79), SPTA1 (30.78% (24/79)), EPB42 (6.33% (5/79)), and SPTB (5.06% (4/79)). Composite genotype was observed in 26.58% (21/79) of patients and included mutations in two or more HS-related genes or mutations in HS-related genes combined with thalassemia or G6PD deficiency. No significant differences in clinical symptoms were found among patients of various genotypes except total bilirubin. Mean reticulocyte volume (MRV) and mean sphered cell volume (MSCV) of the composite genotype were significantly different from other groups. A total of 28 mutation types were found in HS-related genes. Using high-throughput sequencing technology, we also found some cases that had been misdiagnosed. MRV and MSCV are more significant in compound mutations as sensitive determinants of HS. High-throughput sequencing technology can be used to provide a more effective etiological diagnostic method for HS, with high efficiency and specificity.

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来源期刊
Journal of Hematopathology
Journal of Hematopathology HEMATOLOGYPATHOLOGY-PATHOLOGY
CiteScore
0.80
自引率
0.00%
发文量
45
期刊介绍: The Journal of Hematopathology aims at providing pathologists with a special interest in hematopathology with all the information needed to perform modern pathology in evaluating lymphoid tissues and bone marrow. To this end the journal publishes reviews, editorials, comments, original papers, guidelines and protocols, papers on ancillary techniques, and occasional case reports in the fields of the pathology, molecular biology, and clinical features of diseases of the hematopoietic system. The journal is the unique reference point for all pathologists with an interest in hematopathology. Molecular biologists involved in the expanding field of molecular diagnostics and research on lymphomas and leukemia benefit from the journal, too. Furthermore, the journal is of major interest for hematologists dealing with patients suffering from lymphomas, leukemias, and other diseases. The journal is unique in its true international character. Especially in the field of hematopathology it is clear that there are huge geographical variations in incidence of diseases. This is not only locally relevant, but due to globalization, relevant for all those involved in the management of patients.
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