Anti-interleukin 1 treatment in secondary amyloidosis associated with autoinflammatory diseases

Pediatr Nephrol. 2016 Apr;31(4):633-40. doi: 10.1007/s00467-015-3249-5. Epub 2015 Nov 12.

Abstract

Background: Amyloidosis may complicate autoinflammatory diseases (AID). We aimed to evaluate the renal biopsy findings, and clinical and laboratory parameters in patients with AID-associated amyloidosis who have responded to anti-interleukin 1(IL1) treatment.

Methods: Two children with systemic juvenile idiopathic arthritis and one with cryopyrin-associated periodic syndrome diagnosed as having reactive amyloidosis were treated with anti-IL1 drugs. The renal histopathological findings at the time of diagnosis of amyloidosis and after the onset of anti-IL1 were evaluated according to the amyloid scoring/grading system.

Results: The median age of disease onset and diagnosis of amyloidosis were 3 and 12 years, respectively. Anakinra was started in all; however, anakinra caused a local cutaneous reaction in one, thus canakinumab was commenced. Proteinuria improved in all. Control renal biopsies were performed a median of 3 years after the first biopsies. The renal amyloid prognostic score did not improve in patient 1, and progressed in patients 2 and 3. The renal amyloid grade progressed in patient 2.

Conclusions: This is the first series demonstrating progression of renal tissue damage after the improvement of proteinuria with anti-IL 1 in AID-associated amyloidosis. Anti-IL1 drugs are important to prevent further amyloid accumulation; however, new treatment strategies are needed to target the amyloid deposits.

Keywords: Anakinra; Anti-interleukin 1; Autoinflammatory disease; Proteinuria; Renal amyloidosis.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Amyloidosis / diagnosis
  • Amyloidosis / drug therapy*
  • Amyloidosis / immunology
  • Arthritis, Juvenile / diagnosis
  • Arthritis, Juvenile / drug therapy*
  • Arthritis, Juvenile / immunology
  • Biopsy
  • Child
  • Child, Preschool
  • Cryopyrin-Associated Periodic Syndromes / diagnosis
  • Cryopyrin-Associated Periodic Syndromes / drug therapy*
  • Cryopyrin-Associated Periodic Syndromes / immunology
  • Disease Progression
  • Female
  • Humans
  • Immunologic Factors / adverse effects
  • Immunologic Factors / therapeutic use*
  • Interleukin 1 Receptor Antagonist Protein / adverse effects
  • Interleukin 1 Receptor Antagonist Protein / therapeutic use*
  • Kidney / drug effects*
  • Kidney / immunology
  • Kidney / pathology
  • Kidney Diseases / diagnosis
  • Kidney Diseases / drug therapy*
  • Kidney Diseases / immunology
  • Male
  • Proteinuria / drug therapy
  • Proteinuria / immunology
  • Retrospective Studies
  • Time Factors
  • Treatment Outcome

Substances

  • Immunologic Factors
  • Interleukin 1 Receptor Antagonist Protein