Rathmell J C, Goodnow C C
Program in Immunology, Stanford University School of Medicine, CA 94305.
J Immunol. 1994 Sep 15;153(6):2831-42.
Mice homozygous for the lymphoproliferation (lpr) mutation, which disrupts expression of the Fas cell surface molecule, develop an autoimmune syndrome with a spectrum of autoantibodies resembling human SLE. It is not known how the loss of Fas leads to autoantibody production. To study the fate of autoreactive B cells in lpr/lpr mice, C57BL/6 (B6) strain transgenic mice expressing hen egg lysozyme (HEL) as a model autoantigen in soluble or membrane-bound forms and carrying HEL-specific Ig (Ig) transgenes were mated onto the congenic B6-lpr/lpr background. Despite the absence of Fas, elimination of self-reactive lpr/lpr B cells recognizing membrane-bound autoantigen occurred as efficiently as in autoreactive B cells bearing the wild-type (+/+) Fas gene. Functional inactivation of autoreactive B cells binding soluble HEL also occurred normally in most young lpr/lpr animals. Nevertheless, breakdown of B cell tolerance to soluble lysozyme occurred in one of eight young lpr/lpr animals and in four of seven old animals with lymphadenopathy. Interestingly, the presence of the rearranged Ig transgenes markedly delayed the onset of lymphadenopathy. These results demonstrate that Fas is not an essential molecule in the biochemical pathways mediating autoreactive B cell elimination or inactivation. The breakdown of tolerance observed in a considerable fraction of older animals nevertheless confirms that autoantibody production in this model of SLE involves a defect in active censoring of autoreactive B cells. The possible basis for that defect is discussed.
淋巴增殖(lpr)突变的纯合子小鼠会破坏Fas细胞表面分子的表达,从而发展出一种自身免疫综合征,伴有一系列类似于人类系统性红斑狼疮(SLE)的自身抗体。目前尚不清楚Fas的缺失是如何导致自身抗体产生的。为了研究lpr/lpr小鼠中自身反应性B细胞的命运,将表达溶菌酶(HEL)作为模型自身抗原的可溶性或膜结合形式并携带HEL特异性免疫球蛋白(Ig)转基因的C57BL/6(B6)品系转基因小鼠与同基因的B6-lpr/lpr背景进行交配。尽管缺乏Fas,但识别膜结合自身抗原的自身反应性lpr/lpr B细胞的清除与携带野生型(+/+)Fas基因的自身反应性B细胞一样有效。在大多数年轻的lpr/lpr动物中,结合可溶性HEL的自身反应性B细胞的功能失活也正常发生。然而,在八只年轻的lpr/lpr动物中有一只以及七只患有淋巴结病的老年动物中有四只出现了B细胞对可溶性溶菌酶耐受性的破坏。有趣的是,重排的Ig转基因的存在显著延迟了淋巴结病的发作。这些结果表明,Fas在介导自身反应性B细胞清除或失活的生化途径中不是必需分子。然而,在相当一部分老年动物中观察到的耐受性破坏证实,在这个SLE模型中自身抗体的产生涉及自身反应性B细胞主动审查的缺陷。讨论了该缺陷的可能原因。