C3 nephritic factor associated with C3 glomerulopathy in children

Pediatr Nephrol. 2014 Jan;29(1):85-94. doi: 10.1007/s00467-013-2605-6. Epub 2013 Sep 26.

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.

MeSH terms

  • Adolescent
  • Child
  • Child, Preschool
  • Complement C3 Nephritic Factor / metabolism*
  • Female
  • Glomerulonephritis / metabolism
  • Glomerulonephritis / pathology
  • Glomerulonephritis / therapy
  • Glomerulonephritis, Membranoproliferative / metabolism*
  • Glomerulonephritis, Membranoproliferative / pathology*
  • Glomerulonephritis, Membranoproliferative / therapy
  • Humans
  • Infant
  • Male
  • Retrospective Studies

Substances

  • Complement C3 Nephritic Factor