Rudbeck Laboratory, Department of Genetics and Pathology, Uppsala University, Uppsala, Sweden.
Am J Med Genet A. 2010 Oct;152A(10):2595-8. doi: 10.1002/ajmg.a.33604.
Somatic genetic variation in health and disease is poorly explored. Monozygotic (MZ) twins are a suitable model for studies of somatic mosaicism since genetic differences in twins derived from the same zygote represent an irrefutable example of somatic variation. We report the analysis of a pair of generally healthy female MZ twins, discordant for somatic mosaicism for aneuploidy of chromosomes X and Y. Both twins are heterozygous carriers of sickle cell disease mutation. Genotyping of blood DNA from both twins using Illumina Human 610 SNP array revealed a copy number imbalance for chromosome X in a proportion of cells in one twin. Fluorescent in situ hybridization (FISH) analysis confirmed monosomy X (45,X) in 7% of proband nucleated blood cells. Unexpectedly, FISH analysis of cells from the other twin revealed 45,X and 46,XY lineages, both present in 1% of cells. The mechanism behind formation of these aneuploidies suggests several aberrant chromosome segregation events in meiosis and mitoses following conception. Our report contributes to the delineation of the frequency of somatic structural genomic variation in normal MZ twins. These results also illustrate the plasticity of the human genome for tolerating large copy number changes in healthy subjects and show the sensitivity of the Illumina platform for detection of aberrations that are present in a minority of the studied cells.
健康与疾病中的体基因组变异研究甚少。同卵(MZ)双胞胎是研究体细胞镶嵌现象的理想模型,因为来自同一受精卵的双胞胎的遗传差异代表了体细胞变异的确凿例证。我们报告了一对通常健康的女性 MZ 双胞胎的分析结果,这对双胞胎存在 X 染色体和 Y 染色体非整倍体的体细胞镶嵌现象。这对双胞胎均为镰状细胞病突变的杂合子携带者。使用 Illumina Human 610 SNP 阵列对来自双胞胎的血液 DNA 进行基因分型,发现其中一个双胞胎的一部分细胞存在 X 染色体拷贝数失衡。荧光原位杂交(FISH)分析证实了 7%的先证者核血细胞存在单体 X(45,X)。出乎意料的是,对另一个双胞胎细胞的 FISH 分析显示存在 45,X 和 46,XY 谱系,两者均存在于 1%的细胞中。这些非整倍体形成的机制表明,在受精后的减数分裂和有丝分裂中存在多次异常的染色体分离事件。我们的报告有助于描绘正常 MZ 双胞胎中体结构基因组变异的频率。这些结果还说明了人类基因组对健康个体中大量拷贝数变化的耐受性,并且表明了 Illumina 平台在检测存在于少数研究细胞中的异常的敏感性。