Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Apr 6, 2021; 9(10): 2281-2288
Published online Apr 6, 2021. doi: 10.12998/wjcc.v9.i10.2281
Recurrent undifferentiated embryonal sarcoma of the liver in adult patient treated by pembrolizumab: A case report
Xiao-He Yu, Jian Huang, Nai-Jian Ge, Ye-Fa Yang, Jin-Yan Zhao
Xiao-He Yu, Jian Huang, Nai-Jian Ge, Ye-Fa Yang, Department of Radioactive Intervention, Eastern Hepatobiliary Surgery Hospital, The Second Military Medical University, Shanghai 200438, China
Xiao-He Yu, Zhongshan Hospital Qingpu Branch, Fudan University, Shanghai 200092, China
Jin-Yan Zhao, Department of Laboratory Medicine, Shanghai General Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai 200080, China
Author contributions: Yu XH and Huang J were the patient’s interventional radiologists, reviewed the literature and contributed to manuscript drafting; Ge NJ reviewed the literature and contributed to manuscript drafting; Zhao JY performed next generation sequencing and immuno-histological analyses and interpretation and contributed to manuscript drafting; Yang YF made the treatment plan and contributed to manuscript drafting; Zhao JY and Yang YF were responsible for the revision of the manuscript for important intellectual content; All authors issued final approval for the version to be submitted.
Supported by National Natural Science Foundation of China, No. 31971249.
Informed consent statement: Written informed consent was obtained from the patient for publication of this case report.
Conflict-of-interest statement: The authors declared that they have no conflicts of interest regarding this work.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Jin-Yan Zhao, MD, Attending Doctor, Department of Laboratory Medicine, Shanghai General Hospital, Shanghai Jiao Tong University School of Medicine, No. 85 Wujin Road, Shanghai 200080, China. jingy1213@163.com
Received: September 20, 2020
Peer-review started: September 20, 2020
First decision: October 17, 2020
Revised: December 30, 2020
Accepted: January 25, 2021
Article in press: January 25, 2021
Published online: April 6, 2021
Abstract
BACKGROUND

Undifferentiated embryonal sarcoma of the liver (UESL) is a neoplasm that rarely develops in adults. The main treatments for UESL are upfront gross total surgical resection and adjuvant multiagent chemotherapy. Here, we report a case of recurrent UESL in an adult treated with pembrolizumab and discuss a method to identify proper candidates for antibody of programmed cell death protein 1 (anti-PD-1) treatment.

CASE SUMMARY

A 69-year-old woman was admitted for abdominal pain that developed for 1 wk. Computed tomography showed a 16 cm mass in the right lobe of the liver. Right hemihepatectomy and lymphadenectomy were performed, and histological diagnosis was UESL. Six months later, the patient suffered from painless obstructive jaundice, and positron emission tomography-computed tomography revealed multiple metastases. Then, percutaneous transhepatic cholangial drainage was applied to reduce jaundice, and radiofrequency ablation was used to control the lesion near the hepatic hilum. However, the patient suffered from a serious fever caused by the tumor. The patient received treatment with pembrolizumab, and the prescribed dosage was 2 mg/kg every 3 wk. After the seventh dose, positron emission tomography-computed tomography revealed that the multiple metastases had nearly disappeared. Radiologic exam was used to evaluate the disease state, and no new lesions were found. Next-generation sequencing and immunohistology were applied to determine the reason why the patient had such a favorable response to pembrolizumab. Tumor mutation burden, microsatellite instability, and programmed death ligand 1 expression can be combined to predict the effect of PD-1 antibodies. When every one of these biomarkers are detected in a tumor patient, the patient may be a proper candidate for PD-1 antibodies.

CONCLUSION

Anti-PD-1 treatment for tumors needs further research to identify indications and proper biomarkers.

Keywords: Undifferentiated embryonal sarcoma of the liver, Pembrolizumab, Programmed cell death protein 1, Tumor mutation burden, Immunohistology, Case report

Core Tip: Undifferentiated embryonal sarcoma of the liver is a neoplasm rarely diagnosed in adults, which was mainly treated by resection and adjuvant multiagent chemotherapy. Immunotherapy, such as antibody of programmed cell death protein 1, has not been reported to be used to treat this disease. Here we reported a recurrent undifferentiated embryonal sarcoma of the liver case in an adult that was treated by pembrolizumab and discussed the way to find the proper candidates for antibody of programmed cell death protein 1 treatment.