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评估因子VIII多态性短串联重复序列标记物在甲型血友病携带者诊断连锁分析中的应用。

Evaluation of factor VIII polymorphic short tandem repeat markers in linkage analysis for carrier diagnosis of hemophilia A.

作者信息

Shrestha Sabina, Dong Sufang, Li Zuhua, Huang Zhuliang, Zheng Fang

机构信息

Center for Gene Diagnosis, Zhongnan Hospital of Wuhan University, Wuhan, Hubei 430071, P.R. China.

出版信息

Biomed Rep. 2016 Aug;5(2):228-232. doi: 10.3892/br.2016.712. Epub 2016 Jul 4.

Abstract

Hemophilia A (HA) is the most common inherited X-linked recessive bleeding disorder caused by heterogeneous mutations in the factor VIII gene (FVIII). Diagnosis of the carrier is critical for preventing the birth of children affected by this coagulation disorder, which ultimately facilitates its management. Due to the heterogeneous nature of mutations, the large inversions and the complexity of the FVIII gene, direct recognition of the disease-associated mutation in HA is complex. Indirect linkage analysis using highly informative heterozygous polymorphic markers is an alternative method for determining the co-segregation of the mutant gene within a family for carrier detection of HA. The aim of the present study was to perform carrier diagnosis in a family with HA. Rapid multifluorescent polymerase chain reaction (PCR) was performed with six extragenic short tandem repeats (STRs), DXS1073, DXS15, DXS8091, DXS1227, DXS991, DXS993 and one intragenic marker, STR22 for linkage analysis in the HA family. All the STR markers employed in the present study were informative for linkages of pathogenic and healthy haplotypes among family members, particularly STR22, DXS1073 and DXS15. The STR marker, STR22, is within the FVIII gene while the DXS1073 and DXS15 markers are very close to the FVIII gene, where the chances of recombination are comparatively low, and provided the most accurate interpretation analysis, indicating that the proband's sister may have been the HA carrier. Rapid multifluorescent PCR using STR markers and linkage analysis was identified to be a simple method for performing HA carrier diagnosis.

摘要

甲型血友病(HA)是最常见的遗传性X连锁隐性出血性疾病,由凝血因子VIII基因(FVIII)的异质性突变引起。携带者的诊断对于预防受这种凝血障碍影响的儿童出生至关重要,这最终有助于对该疾病的管理。由于突变的异质性、大片段倒位以及FVIII基因的复杂性,直接识别HA中与疾病相关的突变很复杂。使用信息丰富的杂合多态性标记进行间接连锁分析是确定家族中突变基因共分离以检测HA携带者的另一种方法。本研究的目的是对一个患有HA的家族进行携带者诊断。对六个基因外短串联重复序列(STRs)DXS1073、DXS15、DXS8091、DXS1227、DXS991、DXS993以及一个基因内标记STR22进行快速多重荧光聚合酶链反应(PCR),以对该HA家族进行连锁分析。本研究中使用的所有STR标记对于家族成员中致病和健康单倍型的连锁都是有信息价值的,特别是STR22、DXS1073和DXS15。STR标记STR22位于FVIII基因内,而DXS1073和DXS15标记非常靠近FVIII基因,在那里重组的机会相对较低,并提供了最准确的解释分析,表明先证者的妹妹可能是HA携带者。使用STR标记的快速多重荧光PCR和连锁分析被确定为一种进行HA携带者诊断的简单方法。

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