A severe case of atypical hemolytic uremic syndrome associated with pneumococcal infection and T activation treated successfully with plasma exchange

Transfusion. 2008 Nov;48(11):2448-52. doi: 10.1111/j.1537-2995.2008.01871.x. Epub 2008 Jul 30.

Abstract

Background: A severe nondiarrheal form of hemolytic uremic syndrome in children is associated with pneumococcal infection (pHUS). Neuraminidase released by the pneumococci may cleave N-acetylneuraminic acid residues on red blood cells (RBCs), leading to the exposure of the T cryptantigen and polyagglutinability of RBCs, a process known as T activation. Data suggest a pathogenic role of exposed T antigens on glomeruli interacting with naturally occurring anti-T in the development of renal dysfunction in pHUS. By reducing the levels of anti-T and neuraminidase, plasma exchange (PE) may have a role in the treatment of severe cases of pHUS.

Case report: A previously healthy 2-year-old boy presented with acute renal failure, thrombocytopenia, microangiopathic hemolytic anemia, pneumococcal infection, and T activation of RBCs. A diagnosis of pHUS was made. Due to rapid clinical decline, daily single-volume PE with 5 percent albumin replacement was initiated. Infusion of additional plasma was avoided by using only saline-washed RBCs for transfusion. He made a full recovery after 13 PEs and remained well at follow-up 7 months later.

Results: Polyagglutinability of RBCs was shown by mixing patient RBCs with five normal donor sera. The agglutination assays with a panel of lectins confirmed the specificity of exposed T antigen as the cause of polyagglutinability.

Conclusion: The dramatic response seen in this patient suggests that PE utilizing albumin replacement may benefit patients with severe pHUS.

Publication types

  • Case Reports

MeSH terms

  • Anti-Bacterial Agents / therapeutic use
  • Antigens, Tumor-Associated, Carbohydrate / analysis*
  • Bacterial Proteins / metabolism
  • Child, Preschool
  • Combined Modality Therapy
  • Coombs Test
  • Erythrocyte Aggregation
  • Erythrocyte Transfusion
  • Hemagglutination Tests
  • Hemolytic-Uremic Syndrome / blood
  • Hemolytic-Uremic Syndrome / etiology
  • Hemolytic-Uremic Syndrome / therapy*
  • Humans
  • Male
  • Neuraminidase / metabolism
  • Plasma Exchange*
  • Pneumococcal Infections / blood
  • Pneumococcal Infections / complications*
  • Pneumococcal Infections / drug therapy
  • Remission Induction
  • Renal Dialysis
  • Respiration, Artificial
  • Serum Albumin / administration & dosage
  • Streptococcus pneumoniae / enzymology

Substances

  • Anti-Bacterial Agents
  • Antigens, Tumor-Associated, Carbohydrate
  • Bacterial Proteins
  • Serum Albumin
  • Thomsen-Friedenreich antigen
  • Neuraminidase