Homozygous C1q deficiency causes glomerulonephritis associated with multiple apoptotic bodies - PubMed (original) (raw)
Homozygous C1q deficiency causes glomerulonephritis associated with multiple apoptotic bodies
M Botto et al. Nat Genet. 1998 May.
Abstract
The complement system plays a paradoxical role in the development and expression of autoimmunity in humans. The activation of complement in systemic lupus erythematosus (SLE) contributes to tissue injury. In contrast, inherited deficiency of classical pathway components, particularly C1q (ref. 1), is powerfully associated with the development of SLE. This leads to the hypothesis that a physiological action of the early part of the classical pathway protects against the development of SLE (ref. 2) and implies that C1q may play a key role in this respect. C1q-deficient (C1qa-/-) mice were generated by gene targeting and monitored for eight months. C1qa-/- mice had increased mortality and higher titres of autoantibodies, compared with strain-matched controls. Of the C1qa-/- mice, 25% had glomerulonephritis with immune deposits and multiple apoptotic cell bodies. Among mice without glomerulonephritis, there were significantly greater numbers of glomerular apoptotic bodies in C1q-deficient mice compared with controls. The phenotype associated with C1q deficiency was modified by background genes. These findings are compatible with the hypothesis that C1q deficiency causes autoimmunity by impairment of the clearance of apoptotic cells.
Comment in
- The lupus paradox.
Carroll MC. Carroll MC. Nat Genet. 1998 May;19(1):3-4. doi: 10.1038/ng0598-3. Nat Genet. 1998. PMID: 9590274 No abstract available.
Similar articles
- Cutting edge: C1q protects against the development of glomerulonephritis independently of C3 activation.
Mitchell DA, Taylor PR, Cook HT, Moss J, Bygrave AE, Walport MJ, Botto M. Mitchell DA, et al. J Immunol. 1999 May 15;162(10):5676-9. J Immunol. 1999. PMID: 10229798 - C1q and systemic lupus erythematosus.
Walport MJ, Davies KA, Botto M. Walport MJ, et al. Immunobiology. 1998 Aug;199(2):265-85. doi: 10.1016/S0171-2985(98)80032-6. Immunobiology. 1998. PMID: 9777411 Review. - Restoration of C1q levels by bone marrow transplantation attenuates autoimmune disease associated with C1q deficiency in mice.
Cortes-Hernandez J, Fossati-Jimack L, Petry F, Loos M, Izui S, Walport MJ, Cook HT, Botto M. Cortes-Hernandez J, et al. Eur J Immunol. 2004 Dec;34(12):3713-22. doi: 10.1002/eji.200425616. Eur J Immunol. 2004. PMID: 15517607 - C1q knock-out mice for the study of complement deficiency in autoimmune disease.
Botto M. Botto M. Exp Clin Immunogenet. 1998;15(4):231-4. doi: 10.1159/000019076. Exp Clin Immunogenet. 1998. PMID: 10072632 Review. - Hereditary C1q deficiency and systemic lupus erythematosus.
Bowness P, Davies KA, Norsworthy PJ, Athanassiou P, Taylor-Wiedeman J, Borysiewicz LK, Meyer PA, Walport MJ. Bowness P, et al. QJM. 1994 Aug;87(8):455-64. QJM. 1994. PMID: 7922299 Review.
Cited by
- C1q deficiency: identification of a novel missense mutation and treatment with fresh frozen plasma.
Topaloglu R, Taskiran EZ, Tan C, Erman B, Ozaltin F, Sanal O. Topaloglu R, et al. Clin Rheumatol. 2012 Jul;31(7):1123-6. doi: 10.1007/s10067-012-1978-4. Epub 2012 May 11. Clin Rheumatol. 2012. PMID: 22576477 - Advances in lupus genetics.
Niewold TB. Niewold TB. Curr Opin Rheumatol. 2015 Sep;27(5):440-7. doi: 10.1097/BOR.0000000000000205. Curr Opin Rheumatol. 2015. PMID: 26218512 Free PMC article. Review. - Depletion of complement does not impact initiation of xenobiotic-induced autoimmune disease.
Cauvi DM, Toomey CB, Pollard KM. Cauvi DM, et al. Immunology. 2012 Apr;135(4):333-43. doi: 10.1111/j.1365-2567.2011.03545.x. Immunology. 2012. PMID: 22136142 Free PMC article. - Fundamental roles of the innate-like repertoire of natural antibodies in immune homeostasis.
Vas J, Grönwall C, Silverman GJ. Vas J, et al. Front Immunol. 2013 Feb 5;4:4. doi: 10.3389/fimmu.2013.00004. eCollection 2013. Front Immunol. 2013. PMID: 23386848 Free PMC article. - Extracellular vesicles and lupus nephritis - New insights into pathophysiology and clinical implications.
Zhao Y, Wei W, Liu ML. Zhao Y, et al. J Autoimmun. 2020 Dec;115:102540. doi: 10.1016/j.jaut.2020.102540. Epub 2020 Sep 4. J Autoimmun. 2020. PMID: 32893081 Free PMC article. Review.
Publication types
MeSH terms
Substances
LinkOut - more resources
Full Text Sources
Other Literature Sources
Molecular Biology Databases
Miscellaneous