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Background <p>Primary hepatic angiosarcoma (PHA) is an exceptionally rare and aggressive mesenchymal malignancy. Its non-specific clinical manifestations and inconclusive laboratory or imaging findings often lead to delayed diagnosis, typically at advanced stages. The disease carries a dismal prognosis with a median survival of only 2 months, and no standardized therapeutic protocols currently exist.</p> Case Presentation <p>A 59-year-old female diagnosed with diffuse PHA was treated with combination therapy with sintilimab, an anti-programmed cell death protein 1 (PD-1) monoclonal antibody, and anlotinib, a multi-target tyrosine kinase inhibitor (TKI) targeting VEGFR/FGFR pathways. Although the clinical course was marked by biochemical and focal radiographic progression following initial disease control, the patient achieved a notable overall survival of 15 months and remained alive at last contact.</p> Results <p>Serial monitoring revealed stable hepatic function parameters and contained metastatic progression. The patient attained a survival of 15 months with a preserved performance status until the last follow-up.</p> Conclusion <p>This case represents the rare reported instance of significantly prolonged survival in PHA achieved through PD-1 blockade combined with anti-angiogenic therapy. Our findings suggest that the synergistic antitumor effects of immunomodulation and tyrosine kinase inhibition may alter the disease trajectory, providing valuable insights for developing targeted regimens for this treatment-refractory malignancy.</p>

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Combination of Immune Checkpoint Inhibitors and Tyrosine Kinase Inhibitors for the Treatment of Late Stage Diffuse Primary Hepatic Angiosarcoma: a Case Report and Literature Review

  • 期刊论文

  • 发表日期：

    2026年4月7日

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  • Ruolin Lyu,
  • Hongyang Shu,
  • Xiaofan Wu,
  • Yiwen Liu,
  • Ming Wang

摘要

Background

Primary hepatic angiosarcoma (PHA) is an exceptionally rare and aggressive mesenchymal malignancy. Its non-specific clinical manifestations and inconclusive laboratory or imaging findings often lead to delayed diagnosis, typically at advanced stages. The disease carries a dismal prognosis with a median survival of only 2 months, and no standardized therapeutic protocols currently exist.

Case Presentation

A 59-year-old female diagnosed with diffuse PHA was treated with combination therapy with sintilimab, an anti-programmed cell death protein 1 (PD-1) monoclonal antibody, and anlotinib, a multi-target tyrosine kinase inhibitor (TKI) targeting VEGFR/FGFR pathways. Although the clinical course was marked by biochemical and focal radiographic progression following initial disease control, the patient achieved a notable overall survival of 15 months and remained alive at last contact.

Results

Serial monitoring revealed stable hepatic function parameters and contained metastatic progression. The patient attained a survival of 15 months with a preserved performance status until the last follow-up.

Conclusion

This case represents the rare reported instance of significantly prolonged survival in PHA achieved through PD-1 blockade combined with anti-angiogenic therapy. Our findings suggest that the synergistic antitumor effects of immunomodulation and tyrosine kinase inhibition may alter the disease trajectory, providing valuable insights for developing targeted regimens for this treatment-refractory malignancy.