The use of eculizumab in Capnocytophaga canimorsus associated thrombotic microangiopathy: a case report

BMC Infect Dis. 2021 Feb 1;21(1):137. doi: 10.1186/s12879-021-05789-2.

Abstract

Background: The use of complement inhibition is well established for complement mediated thrombotic microangiopathy, but its role in secondary forms of thrombotic microangiopathy is debated. We here present a case of thrombotic microangiopathy triggered by Capnocytophaga canimorsus, illustrating the diagnostic difficulties in discriminating between different thrombotic microangiopathies, and the dilemmas regarding how to treat this disease entity.

Case presentation: A previously healthy 56-year-old woman presented with fever and confusion. She was diagnosed with sepsis from Capnocytophaga canimorsus and thrombotic microangiopathy. Marked activation of both T-cells, endothelium and complement were documented. She was successfully treated with antimicrobial therapy, the complement inhibitor eculizumab and splenectomy. After several weeks, a heterozygote variant in complement factor B was localized, potentially implying the diagnosis of a complement mediated TMA over an isolated infection related TMA.

Conclusions: We discuss the possible interactions between complement activation and other findings in severe infection and argue that complement inhibition proved beneficial to this patient's rapid recovery.

Keywords: Capnocytophaga canimorsus; Case report; Complement; Eculizumab; Thrombotic microangiopathy.

Publication types

  • Case Reports

MeSH terms

  • Antibodies, Monoclonal, Humanized / therapeutic use*
  • Capnocytophaga / pathogenicity*
  • Complement Activation*
  • Complement Inactivating Agents / therapeutic use*
  • Female
  • Gram-Negative Bacterial Infections
  • Humans
  • Middle Aged
  • Norway / epidemiology
  • Sepsis / etiology
  • Thrombotic Microangiopathies / diagnosis*
  • Thrombotic Microangiopathies / drug therapy*

Substances

  • Antibodies, Monoclonal, Humanized
  • Complement Inactivating Agents
  • eculizumab

Supplementary concepts

  • Capnocytophaga canimorsus