Lynch综合征患者亨廷顿基因CAG重复序列大小

S. Gebre-Medhin, K. Dalene Skarping, A. Petersen
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摘要

由于编码DNA错配修复蛋白MLH1、MSH2、MSH6和PMS2的基因存在杂合性种系致病变异,Lynch综合征(LS)患者容易发生癌症。由于受影响基因的第二拷贝失活,LS癌细胞表现出DNA错配修复缺陷和微卫星不稳定。为了研究LS非肿瘤性细胞的微卫星特征,我们测定了MLH1 (n= 11)、MSH2 (n= 9)、MSH6 (n= 7)和非LS对照(n=19) LS患者淋巴细胞DNA中亨廷顿蛋白基因(HTT)微卫星的CAG重复大小。LS的平均重复大小分别为19.55 CAG (MLH1)、19.39 CAG (MSH2)、18.07 CAG (MSH6),而对照组为18.42 CAG。与对照组的3342 CAG相比,LS中CAG重复序列大小的标准差分别为4183 CAG (MLH1)、5089 CAG (MSH2)、3075 CAG (MSH6)。与对照组的27 CAG相比,LS的CAG重复数峰值分别为32 CAG (MLH1)、32 CAG (MSH2)、24 CAG (MSH6)。总的来说,我们的数据表明,在MLH1和MSH2致病变异引起的LS患者中,HTT CAG重复序列的大小往往更大,变化更大。
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Huntingtin gene CAG repeat size in patients with Lynch syndrome
Patients with Lynch syndrome (LS) are prone to cancer due to heterozygous germline pathogenic variants in genes encoding DNA mismatch repair proteins MLH1, MSH2, MSH6 and PMS2. LS cancer cells exhibit deficient DNA mismatch repair and microsatellite instability due somatic inactivation of the second copy of the affected gene. To study microsatellite characteristics in non-neoplastic cells in LS we determined CAG repeat size in the huntingtin gene (HTT) microsatellite in lymphocyte DNA from LS patients with germline pathogenic variants in MLH1 (n = 11), MSH2 (n = 9), MSH6 (n = 7) and non-LS controls (n=19). Mean repeat size in LS was 19,55 CAG (MLH1), 19,39 CAG (MSH2), 18.07 CAG (MSH6), respectively compared to 18,42 CAG in controls. Standard deviation for CAG repeat size in LS was 4,183 CAG (MLH1), 5,089 CAG (MSH2), 3,075 CAG (MSH6), respectively, compared to 3,342 CAG in controls. Peak CAG repeat size in LS was 32 CAG (MLH1), 32 CAG (MSH2), 24 CAG (MSH6), respectively compared to 27 CAG in controls. Collectively, our data indicate that HTT CAG repeat size tends to be larger and more variable in individuals with LS caused by pathogenic variants in MLH1 and MSH2.
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C03 FAN1 coding variants identified in individuals with Huntington’s disease implicate its nuclease activity and dna binding in age at onset C12 HTT repeat instability in family trios in the 100,000 genomes project C08 Knockout of mismatch repair genes MLH1 and MSH3 ablates expansion of the CAG repeat in a human iPSC model of Huntington’s disease C06 The role of FAN1 in HTT cag repeat instability and age at onset in the R6/1 mouse model of Huntington’s disease C10 Haplotype structure analysis of Indian Huntington’s disease patients at HTT gene locus
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