Nath Swapan K, Quintero-Del-Rio Ana I, Kilpatrick Jeff, Feo Lourdes, Ballesteros Maria, Harley John B
Arthritis and Immunology Research Program and Genetic Epidemiology Unit, Oklahoma Medical Research Foundation, Oklahoma City, OK 73104, USA.
Am J Hum Genet. 2004 Jan;74(1):73-82. doi: 10.1086/380913. Epub 2003 Dec 4.
Systemic lupus erythematosus (SLE) is a chronic, complex, and systemic human autoimmune disease, with both an environmental component and a heritable predisposition. Clinical studies, reinforced by epidemiology and genetics, show impressive variation in disease severity, expression, prevalence, and incidence by ethnicity and sex. To identify the novel SLE susceptibility loci, we performed a genomewide scan with 318 markers on 37 multiplex Hispanic families, using a nonparametric penetrance-independent affected-only allele-sharing method. Three chromosomal regions (12q24, 16p13, and 16q12-21) exceeded our predetermined threshold (Zlr>2.32; nominal P<.01) for further evaluation. Suspected linkages at 12q24, 16p13, and 16q12-21 were tested in an independent data set consisting of 92 European American (EA-1) and 55 African American (AA) families. The linkage at 12q24 was replicated in EA-1 (Zlr=3.06; P=.001) but not in AA (Zlr=0.37; P=.35). Although neither the 16p13 nor the 16q12-21 was confirmed in EA-1 or AA, the suggestive linkage (Zlr=3.06; P=.001) at 16q12-21 is sufficient to confirm the significant linkage, reported elsewhere, at this location. The evidence for linkage at 12q24 in the 129 combined (Hispanic and EA-1) families exceeded the threshold for genomewide significance (Zlr=4.39; P=5.7x10-6; nonparametric LOD=4.19). Parametric linkage analyses suggested a low-penetrance, dominant model (LOD=3.72). To confirm the linkage effect at 12q24, we performed linkage analysis in another set of 82 independent European American families (EA-2). The evidence for linkage was confirmed (Zlr=2.11; P=.017). Therefore, our results have detected, established, and confirmed the existence of a novel SLE susceptibility locus at 12q24 (designated "SLEB4") that may cause lupus, especially in Hispanic and European American families.
系统性红斑狼疮(SLE)是一种慢性、复杂的全身性人类自身免疫性疾病,具有环境因素和遗传易感性。临床研究在流行病学和遗传学的支持下表明,该疾病在严重程度、表现形式、患病率和发病率方面因种族和性别存在显著差异。为了确定新的SLE易感基因座,我们采用非参数、不依赖外显率的仅受影响个体等位基因共享方法,对37个西班牙裔多重家庭中的318个标记进行了全基因组扫描。三个染色体区域(12q24、16p13和16q12 - 21)超过了我们预先设定的进一步评估阈值(Zlr>2.32;名义P<0.01)。在由92个欧裔美国人(EA - 1)家庭和55个非裔美国人(AA)家庭组成的独立数据集中,对12q24、16p and 16q12 - 21处的疑似连锁进行了检测。12q24处的连锁在EA - 1中得到复制(Zlr = 3.06;P = 0.001),但在AA中未得到复制(Zlr = 0.37;P = 0.35)。尽管16p13和16q12 - 21在EA - 1或AA中均未得到证实,但16q12 - 至21处的提示性连锁(Zlr = 3.06;P = 0.001)足以证实其他地方报道的该位置的显著连锁。在129个合并的(西班牙裔和EA - 1)家庭中,12q24处的连锁证据超过了全基因组显著性阈值(Zlr = 4.39;P = 5.7×10 - 6;非参数LOD = 4.19)。参数连锁分析提示为低外显率显性模型(LOD = 3.72)。为了证实12q24处的连锁效应,我们在另一组82个独立的欧裔美国人家庭(EA - 2)中进行了连锁分析。连锁证据得到证实(Zlr = 2.11;P = 0.017)。因此,我们的研究结果检测、确立并证实了位于12q24处(命名为“SLEB4”)的一个新的SLE易感基因座的存在,该基因座可能导致狼疮,尤其在西班牙裔和欧裔美国人家庭中。