Division of Laboratory Animal Medicine, University of North Carolina, Chapel Hill, NC, USA.
Mamm Genome. 2014 Apr;25(3-4):95-108. doi: 10.1007/s00335-013-9499-2. Epub 2014 Feb 1.
Inflammatory bowel disease (IBD) is an immune-mediated condition driven by improper responses to intestinal microflora in the context of environmental and genetic background. GWAS in humans have identified many loci associated with IBD, but animal models are valuable for dissecting the underlying molecular mechanisms, characterizing environmental and genetic contributions and developing treatments. Mouse models rely on interventions such as chemical treatment or introduction of an infectious agent to induce disease. Here, we describe a new model for IBD in which the disease develops spontaneously in 20-week-old mice in the absence of known murine pathogens. The model is part of the Collaborative Cross and came to our attention due to a high incidence of rectal prolapse in an incompletely inbred line. Necropsies revealed a profound proliferative colitis with variable degrees of ulceration and vasculitis, splenomegaly and enlarged mesenteric lymph nodes with no discernible anomalies of other organ systems. Phenotypic characterization of the CC011/Unc mice with homozygosity ranging from 94.1 to 99.8% suggested that the trait was fixed and acted recessively in crosses to the colitis-resistant C57BL/6J inbred strain. Using a QTL approach, we identified four loci, Ccc1, Ccc2, Ccc3 and Ccc4 on chromosomes 12, 14, 1 and 8 that collectively explain 27.7% of the phenotypic variation. Surprisingly, we also found that minute levels of residual heterozygosity in CC011/Unc have significant impact on the phenotype. This work demonstrates the utility of the CC as a source of models of human disease that arises through new combinations of alleles at susceptibility loci.
炎症性肠病(IBD)是一种免疫介导的疾病,其发病机制是在环境和遗传背景下,对肠道微生物群产生异常反应。人类全基因组关联研究已经确定了许多与 IBD 相关的基因座,但动物模型对于剖析潜在的分子机制、描述环境和遗传因素的作用以及开发治疗方法非常有价值。小鼠模型依赖于化学处理或引入感染因子等干预措施来诱导疾病。在这里,我们描述了一种新的 IBD 模型,该模型在没有已知的小鼠病原体的情况下,20 周龄的小鼠会自发发病。该模型是协作交叉的一部分,由于不完全近交系中直肠脱垂的发病率很高,引起了我们的注意。尸检显示严重的增生性结肠炎,伴有不同程度的溃疡和血管炎、脾肿大和肠系膜淋巴结肿大,但其他器官系统没有明显异常。对 CC011/Unc 小鼠(杂合度为 94.1%至 99.8%)进行表型特征分析表明,该性状是固定的,在与结肠炎抗性 C57BL/6J 近交系杂交时表现为隐性。使用 QTL 方法,我们在 12、14、1 和 8 号染色体上确定了四个基因座 Ccc1、Ccc2、Ccc3 和 Ccc4,它们共同解释了 27.7%的表型变异。令人惊讶的是,我们还发现 CC011/Unc 中微小水平的残余杂合性对表型有显著影响。这项工作证明了 CC 作为人类疾病模型来源的实用性,这些模型是通过易感基因座中等位基因的新组合产生的。