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补体第八成分缺乏与系统性红斑狼疮样疾病相关

Absence of the eighth component of complement in association with systemic lupus erythematosus-like disease.

作者信息

Jasin H E

出版信息

J Clin Invest. 1977 Sep;60(3):709-15. doi: 10.1172/JCI108823.

Abstract

A 56-yr-old black woman with absence of the eighth component of complement and a disease compatible with systemic lupus erythematosus is described. Her disease is characterized by the presence of photosensitive "malar" rash, alopecia, polyarthritis, and nephrotic syndrome. Hemolytic and immunochemical assays of the serum complement components were normal, except for C8 which was undetectable. Hemolytic activity could be restored to normal by the addition of functionally pure C8. In vitro tests to investigate the functional integrity of the classical and alternative pathways indicated that the functions mediated by activation of C3 and C5 were intact whereas heatlabile bactericidal activity was totally absent. Addition of C8 restored this function to normal levels. One of two brothers of the proband was shown to have serum C8 levels approaching 50% of normal indicating the hereditary nature of the defect. HLA typing studies showed that the normal brother had identical haplotypes to the proband while the proposed heterozygous brother only shared one haplotype with the patient, suggesting that the gene controlling the C8 defect was not closely linked to the major histocompatibility complex. If the association of a connective tissue disease and absence of a terminal component of complement is not coincidental, these results suggest that C8 deficiency may be associated with a subtle defect in the defense mechanisms to viral infection leading to viral persistance and perhaps to diseases such as systemic lupus erythematosus where chronic viral infections have been implicated.

摘要

本文描述了一位56岁的黑人女性,其缺乏补体第八成分,患有与系统性红斑狼疮相符的疾病。她的疾病特征为存在光敏性“蝶形”皮疹、脱发、多关节炎和肾病综合征。血清补体成分的溶血和免疫化学检测均正常,但C8检测不到。通过添加功能纯的C8可使溶血活性恢复正常。用于研究经典途径和替代途径功能完整性的体外试验表明,由C3和C5激活介导的功能是完整的,而热不稳定杀菌活性完全缺失。添加C8可使该功能恢复到正常水平。先证者的两个兄弟中有一个血清C8水平接近正常的50%,表明该缺陷具有遗传性。HLA分型研究表明,正常兄弟与先证者具有相同的单倍型,而推测为杂合子的兄弟仅与患者共享一个单倍型,这表明控制C8缺陷的基因与主要组织相容性复合体没有紧密连锁。如果结缔组织疾病与补体终末成分缺乏之间的关联并非偶然,这些结果提示C8缺乏可能与病毒感染防御机制中的细微缺陷有关,导致病毒持续存在,或许还与诸如系统性红斑狼疮这类涉及慢性病毒感染的疾病有关。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/33a4/372416/a66e0a89e963/jcinvest00657-0212-a.jpg

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