Sellick Gabrielle S, Goldin Lynn R, Wild Ruth W, Slager Susan L, Ressenti Laura, Strom Sara S, Dyer Martin J S, Mauro Francesca R, Marti Gerald E, Fuller Stephen, Lyttelton Matthew, Kipps Thomas J, Keating Michael J, Call Timothy G, Catovsky Daniel, Caporaso Neil, Houlston Richard S
Section of Cancer Genetics, Institute of Cancer Research, 15 Cotswold Road, Sutton, Surrey, UK.
Blood. 2007 Nov 1;110(9):3326-33. doi: 10.1182/blood-2007-05-091561. Epub 2007 Aug 8.
Chronic lymphocytic leukemia (CLL) and other B-cell lymphoproliferative disorders display familial aggregation. To identify a susceptibility gene for CLL, we assembled families from the major European (ICLLC) and American (GEC) consortia to conduct a genome-wide linkage analysis of 101 new CLL pedigrees using a high-density single nucleotide polymorphism (SNP) array and combined the results with data from our previously reported analysis of 105 families. Here, we report on the combined analysis of the 206 families. Multipoint linkage analyses were undertaken using both nonparametric (model-free) and parametric (model-based) methods. After the removal of high linkage disequilibrium SNPs, we obtained a maximum nonparametric linkage (NPL) score of 3.02 (P = .001) on chromosome 2q21.2. The same genomic position also yielded the highest multipoint heterogeneity LOD (HLOD) score under a common recessive model of disease susceptibility (HLOD = 3.11; P = 7.7 x 10(-5)), which was significant at the genome-wide level. In addition, 2 other chromosomal positions, 6p22.1 (corresponding to the major histocompatibility locus) and 18q21.1, displayed HLOD scores higher than 2.1 (P < .002). None of the regions coincided with areas of common chromosomal abnormalities frequently observed in CLL. These findings provide direct evidence for Mendelian predisposition to CLL and evidence for the location of disease loci.
慢性淋巴细胞白血病(CLL)和其他B细胞淋巴增殖性疾病呈现家族聚集性。为了鉴定CLL的一个易感基因,我们整合了来自欧洲主要联盟(ICLLC)和美国联盟(GEC)的家族,使用高密度单核苷酸多态性(SNP)阵列对101个新的CLL家系进行全基因组连锁分析,并将结果与我们之前报道的对105个家系的分析数据相结合。在此,我们报告对这206个家系的联合分析。使用非参数(无模型)和参数(基于模型)方法进行多点连锁分析。去除高度连锁不平衡的SNP后,我们在2号染色体2q21.2处获得了最大非参数连锁(NPL)分数3.02(P = 0.001)。在疾病易感性的常见隐性模型下,相同的基因组位置也产生了最高的多点异质性LOD(HLOD)分数(HLOD = 3.11;P = 7.7×10⁻⁵),在全基因组水平上具有显著性。此外,另外2个染色体位置,6p22.1(对应主要组织相容性位点)和18q21.1,显示HLOD分数高于2.1(P < 0.002)。这些区域均与CLL中常见的染色体异常区域不重合。这些发现为CLL的孟德尔易感性提供了直接证据,并为疾病基因座的定位提供了证据。