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May 15, 2026IJU Case Reports0 citationsOpen Access

Pseudo‐Progression Observed During Immune Checkpoint Inhibitor Therapy for Advanced Adrenocortical Carcinoma

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MSMoeto ShimodaSOShinji OhtakeTATetsuya Aoki

Key Result

Pembrolizumab therapy in a patient with advanced adrenocortical carcinoma and high tumor mutational burden (27.1/Mb) resulted in transient tumor shrinkage before death at 9 months.

Key Points

  • To explore the effectiveness of immune checkpoint inhibitors in advanced adrenocortical carcinoma and assess the significance of tumor mutational burden.
  • Reported a case of a 49-year-old man with advanced adrenocortical carcinoma.
  • Treated with EDP-M chemotherapy followed by pembrolizumab due to high tumor mutational burden.
  • Evaluated treatment response using immune RECIST criteria.
  • Despite suspected pseudo-progression, transient tumor shrinkage was observed.
  • The patient passed away 9 months after diagnosis due to liver failure.
  • Findings suggest immune checkpoint inhibitors could have a therapeutic role in advanced ACC.

Study Design

Type

Case Report (n=1)

Structured PICO

P
Population
1 49-year-old man with advanced adrenocortical carcinoma (ACC) and pulmonary metastasis, with high tumor mutational burden (TMB, 27.1/Mb)
I
Intervention
EDP-M chemotherapy followed by pembrolizumab
O
Outcome
Tumor response and survivalhard clinical

Pembrolizumab may have therapeutic activity in advanced adrenocortical carcinoma with high tumor mutational burden, though pseudo-progression can complicate response evaluation.

Abstract

ABSTRACT Introduction Adrenocortical carcinoma (ACC) is a rare and aggressive malignancy. Recent studies suggest that immune checkpoint inhibitors exhibit antitumor activity in selected ACC patients. Case Presentation We report a case of a 49‐year‐old man with advanced ACC and pulmonary metastasis treated with EDP‐M chemotherapy, followed by pembrolizumab based on high tumor mutational burden (TMB, 27.1/Mb). Despite suspected pseudo‐progression, transient tumor shrinkage was observed. However, the patient died of liver failure 9 months after diagnosis. Conclusion This case suggests a therapeutic role for ICIs in ACC. Moreover, it highlights the importance of TMB and immune RECIST criteria in evaluating treatment response.

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Cite This Study

Shimoda et al. (2026) conducted a case report in Advanced adrenocortical carcinoma (n=1). Pembrolizumab was evaluated. Pembrolizumab therapy in a patient with advanced adrenocortical carcinoma and high tumor mutational burden (27.1/Mb) resulted in transient tumor shrinkage before death at 9 months.

synapsesocial.com/papers/6a06b86ae7dec685947aadc3https://doi.org/10.1002/iju5.70192
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