C3 nephritic factor associated with C3 glomerulopathy in children View Full Text


Ontology type: schema:ScholarlyArticle     


Article Info

DATE

2014-01

AUTHORS

Camille Nicolas, Vincent Vuiblet, Veronique Baudouin, Marie-Alice Macher, Isabele Vrillon, Nathalie Biebuyck-Gouge, Maud Dehennault, Sophie Gié, Denis Morin, Hubert Nivet, François Nobili, Tim Ulinski, Bruno Ranchin, Maria Chiarra Marinozzi, Stéphanie Ngo, Véronique Frémeaux-Bacchi, Christine Pietrement

ABSTRACT

BACKGROUND: C3 glomerulopathy (C3G) is characterized by predominant C3 deposits in glomeruli and dysregulation of the alternative pathway of complement. Half of C3G patients have a C3 nephritic factor (C3NeF). C3G incorporated entities with a range of features on microscopy including dense deposit diseases (DDD) and C3 glomerulonephritis (C3GN). The aim of this work was to study children cases of C3G associated with C3NeF. METHODS: We reviewed 18 cases of C3G with a childhood onset associated with C3NeF without identified mutations in CFH, CFI, and MCP genes. RESULTS: Clinical histories started with recurrent hematuria for seven patients, nephrotic syndrome for four, acute post-infectious glomerulonephritis for three and acute renal failure for four. Twelve patients had a low C3 at first investigation. Kidney biopsy showed ten C3GN and eight DDD. Twenty-three percent of the patients tested presented elevated sC5b9. Seven patients relapsed 3 to 6 years after the onset. At the end of follow-up, two patients were under dialysis, 11 had a persistent proteinuria, five had none; four patients did not follow any treatment. Steroids were first used in 80 % of cases. CONCLUSIONS: C3NeF associated C3G has a heterogeneous presentation and outcome. Anti-proteinuric agents may control the disease during follow-up, even after nephrotic syndrome at the onset. The efficiency of immunosuppressive therapy remains questionable. More... »

PAGES

85-94

Identifiers

URI

http://scigraph.springernature.com/pub.10.1007/s00467-013-2605-6

DOI

http://dx.doi.org/10.1007/s00467-013-2605-6

DIMENSIONS

https://app.dimensions.ai/details/publication/pub.1041761797

PUBMED

https://www.ncbi.nlm.nih.gov/pubmed/24068526


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