Eculizumab in atypical haemolytic uraemic syndrome with severe cardiac and neurological involvement

Pediatr Nephrol. 2014 Jun;29(6):1103-6. doi: 10.1007/s00467-013-2709-z. Epub 2013 Dec 8.

Abstract

Background: Atypical haemolytic uraemic syndrome (aHUS) is a rare disorder usually caused by dysregulation of the alternative complement pathway. Uncontrolled complement activation results in systemic complement-mediated thrombotic microangiopathy (TMA) and subsequent multi-organ damage. The two most common extrarenal complications comprise neurological and cardiovascular involvement. Eculizumab, a humanised anti-C5 monoclonal antibody, has recently been introduced as a therapy for this condition.

Case-diagnosis/treatment: A 19-month-old child suffering from aHUS with severe neurological involvement, dilated cardiomyopathy and renal impairment requiring dialysis received eculizumab as first-line treatment, initiated within 12 h of admission, resulting in significant improvements in her neurological state and normalisation of cardiac and renal function. These positive outcomes have been sustained with fortnightly eculizumab therapy (at the time of writing, on-going for 1 year). No further complications of TMA have occurred.

Conclusion: Severe cardiac involvement in a child with aHUS is an important indication for prompt, first-line treatment with eculizumab, resulting in rapid normalisation of cardiac function.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Antibodies, Monoclonal, Humanized / therapeutic use*
  • Atypical Hemolytic Uremic Syndrome / complications*
  • Atypical Hemolytic Uremic Syndrome / drug therapy*
  • Cardiomyopathy, Dilated / etiology
  • Female
  • Humans
  • Infant
  • Renal Dialysis
  • Renal Insufficiency / etiology
  • Seizures / etiology

Substances

  • Antibodies, Monoclonal, Humanized
  • eculizumab