Xing Chao, Sestak Andrea L, Kelly Jennifer A, Nguyen Kim L, Bruner Gail R, Harley John B, Gray-McGuire Courtney
Division of Genetic and Molecular Epidemiology, Department of Biostatistics and Epidemiology, Case Western Reserve University, Wolstein Research Building Rm 1312, 10900 Euclid Avenue, Cleveland, OH 44106-7281, USA.
Hum Genet. 2007 Jan;120(5):623-31. doi: 10.1007/s00439-006-0248-4. Epub 2006 Sep 16.
Systemic lupus erythematosus (SLE) is a systemic autoimmune disease characterized by both population and phenotypic heterogeneity. Our group previously identified linkage to SLE at 4p16 in European Americans (EA). In the present study we replicate this linkage effect in a new cohort of 76 EA families multiplex for SLE by model-free linkage analysis. Using densely spaced microsatellite markers in the linkage region, we have localized the potential SLE susceptibility gene(s) to be telomeric to the marker D4S2928 by haplotype construction. In addition, marker D4S394 showed marginal evidence of linkage disequilibrium with the putative disease locus by the transmission disequilibrium test and significant evidence of association using a family-based association approach as implemented in the program ASSOC. We also performed both two-point and multipoint model-based analyses to characterize the genetic model of the potential SLE susceptibility gene(s), and the lod scores both maximized under a recessive model with penetrances of 0.8. Finally, we performed a genome-wide scan of the total 153 EA pedigrees and evaluated the possibility of interaction between linkage signals at 4p16 and other regions in the genome. Fourteen regions on 11 chromosomes (1q24, 1q42, 2p11, 2q32, 3p14.2, 4p16, 5p15, 7p21, 8p22, 10q22, 12p11, 12q24, 14q12, 19q13) showed evidence of linkage, among which, signals at 2p11, 12q24 and 19q13 also showed evidence of interaction with that at 4p16. These results provide important additional information about the SLE linkage effect at 4p16 and offer a unique approach to uncovering susceptibility loci involved in complex human diseases.
系统性红斑狼疮(SLE)是一种全身性自身免疫性疾病,具有人群和表型异质性。我们的研究小组之前在欧洲裔美国人(EA)中发现4p16与SLE存在连锁关系。在本研究中,我们通过无模型连锁分析,在一个新的由76个SLE多发家系组成的欧洲裔美国人队列中重复了这种连锁效应。在连锁区域使用密集间隔的微卫星标记,我们通过单倍型构建将潜在的SLE易感基因定位到标记D4S2928的端粒侧。此外,通过传递不平衡检验,标记D4S394显示出与假定疾病位点存在连锁不平衡的边缘证据,并且使用程序ASSOC中实施的基于家系的关联方法显示出显著的关联证据。我们还进行了两点和多点基于模型的分析,以表征潜在的SLE易感基因的遗传模型,在隐性模型下,外显率为0.8时,对数优势分数均达到最大值。最后,我们对总共153个欧洲裔美国人谱系进行了全基因组扫描,并评估了4p16处的连锁信号与基因组中其他区域之间相互作用的可能性。11条染色体上的14个区域(1q24、1q42、2p11、2q32、3p14.2、4p16、5p15、7p21、8p22、10q22、12p11、12q24、14q12、19q13)显示出连锁证据,其中,2p11、12q24和19q13处的信号也显示出与4p16处的信号存在相互作用的证据。这些结果为4p16处的SLE连锁效应提供了重要的补充信息,并为揭示复杂人类疾病中涉及的易感基因座提供了独特的方法。