Gibson W T, Walter M A, Ahmed A R, Alper C A, Cox D W
Research Institute, Hospital for Sick Children, Toronto, Ontario, Canada.
Hum Genet. 1994 Dec;94(6):675-83. doi: 10.1007/BF00206963.
Genes involved in the immune response are generally encoded from a complex cluster of gene segments. Studies of the association of diseases with such genes require well-defined genetic markers throughout the selected region. A set of 15 polymorphic loci that span 1500 kb of the immunoglobulin heavy chain (IGH) complex, 8 in the variable (VH) region and 7 in the constant (CH) region, were selected for the study of disease association. We present a protocol for the use of multiple immunoglobulin heavy chain (IGH) polymorphisms for general application in disease association studies. No microsatellite repeat markers are available for this region. To demonstrate the applicability of this approach, we have examined these IGH polymorphisms in families with individuals affected with pemphigus vulgaris (PV), an autoimmune dermatologic disease. Allele frequencies in 12 patients with PV were compared with those found in their spouses, and with those in a white Canadian control population. A significant difference was found between PV patients and both control groups for the presence of the VH gene VH3f-R4, and possibly for the absence of VH3f-R3, suggesting the possibility of susceptibility factors in these regions. Examination of the frequencies of the IGH region C gamma-haplotypes of PV patients indicated that, while the patients did not differ significantly from their spouses (chi 2 = 1.79), both groups were found to differ significantly from the white Canadian control group (chi 2 = 10.10), emphasizing the importance of matching the ethnic background of controls with that of the patient test group in disease association studies. Unexpectedly, two patients had large deletions of genes in the IGH constant region, which could play a role in the development of PV and require further investigation.
参与免疫反应的基因通常由一组复杂的基因片段编码。研究疾病与这类基因的关联需要在选定区域内有明确的遗传标记。为了研究疾病关联,我们选择了一组15个多态性位点,它们跨越1500 kb的免疫球蛋白重链(IGH)复合体,其中8个位于可变(VH)区域,7个位于恒定(CH)区域。我们提出了一种使用多个免疫球蛋白重链(IGH)多态性的方案,以便在疾病关联研究中普遍应用。该区域没有微卫星重复标记。为了证明这种方法的适用性,我们在患有寻常型天疱疮(PV)(一种自身免疫性皮肤病)的个体的家庭中检测了这些IGH多态性。将12例PV患者的等位基因频率与其配偶的等位基因频率以及加拿大白人对照组的等位基因频率进行了比较。在PV患者与两个对照组之间,发现VH基因VH3f-R4的存在以及可能VH3f-R3的缺失存在显著差异,这表明这些区域可能存在易感因素。对PV患者IGH区域Cγ单倍型频率的检测表明,虽然患者与其配偶之间没有显著差异(χ2 = 1.79),但两组均与加拿大白人对照组存在显著差异(χ2 = 10.10),这强调了在疾病关联研究中使对照组的种族背景与患者测试组的种族背景相匹配的重要性。出乎意料的是,两名患者在IGH恒定区域存在基因大片段缺失,这可能在PV的发病过程中起作用,需要进一步研究。