Case report: Fatal overwhelming post-splenectomy infection in a patient with metastatic angiosarcoma treated with immunotherapy

Front Immunol. 2024 May 8:15:1366271. doi: 10.3389/fimmu.2024.1366271. eCollection 2024.

Abstract

A patient in his 40s with splenic angiosarcoma metastatic to the liver underwent splenectomy, chemotherapy, and partial hepatectomy before being treated on a clinical trial with CTLA4 and PD1 inhibitors. He had received pneumococcal and meningococcal vaccines post-splenectomy. On week 10, he developed grade 3 immune-related colitis, successfully treated with the anti-tumor necrosis factor-alpha inhibitor infliximab and steroids. After 4 cycles of treatment, scans showed partial response. He resumed anti-PD1 therapy, and 6 hours after the second dose of anti-PD1 he presented to the emergency room with hematemesis, hematochezia, hypotension, fever, and oxygen desaturation. Laboratory tests demonstrated acute renal failure and septicemia (Streptococcus pneumoniae). He died 12 hours after the anti-PD1 infusion from overwhelming post-splenectomy infection (OPSI). Autopsy demonstrated non-viable liver tumors among other findings. In conclusion, patients undergoing immunotherapy and with prior history of asplenia should be monitored closely for OPSI as they may be at increased risk.

Keywords: CTLA4; OPSI; immuno-therapy; immunology; infection; splenectomy.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • CTLA-4 Antigen / antagonists & inhibitors
  • Fatal Outcome
  • Hemangiosarcoma* / therapy
  • Humans
  • Immune Checkpoint Inhibitors / adverse effects
  • Immune Checkpoint Inhibitors / therapeutic use
  • Immunotherapy / adverse effects
  • Immunotherapy / methods
  • Liver Neoplasms* / secondary
  • Liver Neoplasms* / therapy
  • Male
  • Pneumococcal Infections / etiology
  • Programmed Cell Death 1 Receptor / antagonists & inhibitors
  • Splenectomy* / adverse effects
  • Splenic Neoplasms* / secondary
  • Splenic Neoplasms* / therapy

Substances

  • Immune Checkpoint Inhibitors
  • Programmed Cell Death 1 Receptor
  • CTLA-4 Antigen

Grants and funding

The author(s) declare financial support was received for the research, authorship, and/or publication of this article. This work was supported in part by the National Institutes of Health/National Cancer Institute award number P30 CA016672 (University of Texas MD Anderson Cancer Center). This work was also supported by the Mr. and Mrs. Steven McKenzie’s Endowment and donor funds from Jamie’s Hope and Mrs. and Mr. James Ritter for Dr. Tsimberidou’s Personalized Medicine Program (Initiative for Molecular Profiling and Advanced Cancer Therapy, IMPACT). The funders had no role in the study design, data collection and analysis, decision to publish, or preparation of the manuscript.