Secondary hemophagocytic syndrome after autologous hematopoietic cell transplant and immune therapy for neuroblastoma

Pediatr Blood Cancer. 2019 Nov;66(11):e27964. doi: 10.1002/pbc.27964. Epub 2019 Aug 12.

Abstract

Secondary hemophagocytic syndrome (HPS) has been described after autologous hematopoietic cell transplant (AutoHCT). We report two cases of secondary HPS after novel consolidation therapy for high-risk neuroblastoma as part of an institutional phase 2 trial incorporating immunotherapy into a "standard" AutoHCT regimen. Both patients developed liver dysfunction beyond expected course of hepatic veno-occlusive disease, coagulopathy, hyperferritinemia, and when evaluated, elevated soluble interleukin-2 receptor and hemophagocytosis. These cases highlight the need for clinicians to have a high index of suspicion for immune-related complications in patients receiving immune therapies.

Keywords: autologous hematopoietic cell transplant; hemophagocytic syndrome; immunotherapy; neuroblastoma.

Publication types

  • Case Reports

MeSH terms

  • Antineoplastic Agents, Immunological / adverse effects*
  • Antineoplastic Agents, Immunological / therapeutic use
  • Antineoplastic Combined Chemotherapy Protocols / adverse effects
  • Antineoplastic Combined Chemotherapy Protocols / therapeutic use
  • Busulfan / administration & dosage
  • Busulfan / adverse effects
  • Child, Preschool
  • Ferritins / blood
  • Hepatic Veno-Occlusive Disease / etiology
  • Humans
  • Immunotherapy / adverse effects*
  • Immunotherapy, Adoptive / adverse effects*
  • Infant
  • Killer Cells, Natural / transplantation*
  • Liver Failure / etiology*
  • Liver Failure / therapy
  • Lymphohistiocytosis, Hemophagocytic / etiology*
  • Lymphohistiocytosis, Hemophagocytic / immunology
  • Male
  • Melphalan / administration & dosage
  • Melphalan / adverse effects
  • Neuroblastoma / drug therapy
  • Neuroblastoma / therapy*
  • Peripheral Blood Stem Cell Transplantation / adverse effects*
  • Systemic Inflammatory Response Syndrome / etiology*
  • Systemic Inflammatory Response Syndrome / therapy
  • Transplantation Conditioning / adverse effects

Substances

  • Antineoplastic Agents, Immunological
  • Ferritins
  • Busulfan
  • Melphalan