Role of eculizumab in a pediatric refractory gemcitabine-induced thrombotic microangiopathy: a case report

J Med Case Rep. 2017 Jul 27;11(1):209. doi: 10.1186/s13256-017-1373-5.

Abstract

Background: The incidence of gemcitabine-induced hemolytic uremic syndrome has already been described in adults. Several approaches have been employed in the treatment of gemcitabine-induced hemolytic uremic syndrome with different outcomes. One of the most promising agents is eculizumab, which is a monoclonal antibody directed against C5 complement protein.

Case presentation: We reported the case of a 3-year-old white boy with medulloblastoma who underwent high-dose chemotherapy and craniospinal irradiation. Afterwards he started maintenance chemotherapy with gemcitabine and oxaliplatin. After five courses he presented a progressive clinical worsening, which resulted in a systemic thrombotic microangiopathy. Initially he was treated with rituximab without clinical improvement. Therefore he started therapy with repeated cycles of eculizumab. After seven infusions he showed a gradual improvement and finally a complete remission of gemcitabine-induced hemolytic uremic syndrome.

Conclusions: Eculizumab prevents serious complement-mediated vascular damage for chemotherapy-induced thrombotic microangiopathy in pediatric cases.

Keywords: Brain tumors; Case report; Eculizumab; Gemcitabine; Medulloblastoma; Microangiopathy; Radiotherapy.

Publication types

  • Case Reports

MeSH terms

  • Antibodies, Monoclonal, Humanized / administration & dosage*
  • Antimetabolites, Antineoplastic / adverse effects*
  • Cerebellar Neoplasms / drug therapy
  • Child, Preschool
  • Complement Inactivating Agents / administration & dosage*
  • Deoxycytidine / adverse effects
  • Deoxycytidine / analogs & derivatives*
  • Fatal Outcome
  • Gemcitabine
  • Hemolytic-Uremic Syndrome / chemically induced
  • Hemolytic-Uremic Syndrome / diagnosis
  • Hemolytic-Uremic Syndrome / drug therapy*
  • Humans
  • Male
  • Medulloblastoma / drug therapy

Substances

  • Antibodies, Monoclonal, Humanized
  • Antimetabolites, Antineoplastic
  • Complement Inactivating Agents
  • Deoxycytidine
  • eculizumab
  • Gemcitabine