A case of C3 glomerulonephritis successfully treated with eculizumab View Full Text


Ontology type: schema:ScholarlyArticle     


Article Info

DATE

2015-03-22

AUTHORS

Alexis Payette, Natalie Patey, Marie-Agnès Dragon-Durey, Véronique Frémeaux-Bacchi, Françoise Le Deist, Anne-Laure Lapeyraque

ABSTRACT

BackgroundC3 glomerulonephritis (C3GN) is a rare form of glomerulopathy that is characterized by predominant C3 deposits. Eculizumab, a humanized monoclonal C5 antibody, has recently emerged as a treatment option for C3GN. We report a C3GN patient successfully treated with eculizumab.Case diagnosis/treatmentA 5-year-old boy who presented with proteinuria, hematuria, high ASO titers, and low C3 levels was initially diagnosed with post-streptococcal GN. His first kidney biopsy confirmed this diagnosis, but complement investigations identified three alternative pathway dysregulation factors: C3 nephritic factor, complement factor I heterozygous mutation (I398L), and anti-factor H autoantibodies (4,500 AU/ml). A second biopsy performed 11 months after initial presentation (nephrotic range proteinuria) showed a C3GN suggestive of isolated C3 deposits.Despite the use of intensive immunosuppressive therapy (rituximab, corticosteroids, mycophenolate), nephrotic-range proteinuria persisted and a third kidney biopsy showed the same C3GN pattern with more endocapillary proliferation. The serum C5b-9 level was elevated. Eculizumab was initiated and resulted in a significant decline of proteinuria (5.3 to 1.3 g/day) and an improvement in pathologic features. A transient interruption of eculizumab resulted in a rapid rise in proteinuria to 9.3 g/day, which decreased to 0.8 g/day after resumption of treatment.ConclusionsThe administration of anti-C5 antibodies may represent a valuable therapeutic option in patients with C3GN. More... »

PAGES

1033-1037

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URI

http://scigraph.springernature.com/pub.10.1007/s00467-015-3061-2

DOI

http://dx.doi.org/10.1007/s00467-015-3061-2

DIMENSIONS

https://app.dimensions.ai/details/publication/pub.1052803881

PUBMED

https://www.ncbi.nlm.nih.gov/pubmed/25796589


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