PMID- 32508620 OWN - NLM STAT- PubMed-not-MEDLINE LR - 20200928 IS - 1662-6575 (Print) IS - 1662-6575 (Electronic) IS - 1662-6575 (Linking) VI - 13 IP - 1 DP - 2020 Jan-Apr TI - Checkpoint Inhibition Causing Complete Remission of Metastatic Combined Hepatocellular-Cholangiocarcinoma after Hepatic Resection. PG - 478-484 LID - 10.1159/000507320 [doi] AB - Combined hepatocellular-cholangiocarcinoma (CHC) is a rare type of primary liver cancer, speculated to arise from hepatic progenitor cells, and with a worse prognosis than hepatocellular carcinoma (HCC). Serum alpha-fetoprotein (AFP) levels may be one prognostic factor. It has been suggested that checkpoint inhibition might be useful in the treatment of HCC where there is an increased expression of PD-1 and PD-L1 in the microenvironment. Its effect on CHC is unknown. We report a case with a large CHC, which was radically resected, but the 53-year-old female patient subsequently developed pulmonary metastases. Histology demonstrated low-differentiated CHC without microsatellite instability. Treatment with sorafenib was started but was stopped due to angioedema. Under subsequent gemcitabine/cisplatin treatment, the metastatic disease progressed with rising AFP levels. A third-line treatment with pembrolizumab was then started, 2 mg/kg b.w. i.v. every third week for 6 months. This resulted in a radiologically complete remission of the pulmonary metastases and AFP levels were normalized (<10 mug/L) from a level of 1,790 mug/L before treatment. The patient developed immune-related adverse events (AEs) including diarrhea and hepatitis. These AEs were successfully treated with prednisolone and mycophenolate mofetil, and they were eventually resolved. There are no signs of cancer recurrence neither in the liver nor in the lungs at 33 months after the start of the checkpoint inhibition treatment, and the patient is doing well. Further study is urgently needed on the role of checkpoint inhibition therapy in liver cancer. CI - Copyright (c) 2020 by S. Karger AG, Basel. FAU - Rizell, Magnus AU - Rizell M AD - Transplant Institute, Sahlgrenska University Hospital, Gothenburg, Sweden. FAU - Aberg, Fredrik AU - Aberg F AD - Transplant Institute, Sahlgrenska University Hospital, Gothenburg, Sweden. FAU - Perman, Mats AU - Perman M AD - Department of Oncology, Sahlgrenska University Hospital, Gothenburg, Sweden. FAU - Ny, Lars AU - Ny L AD - Department of Oncology, Sahlgrenska University Hospital, Gothenburg, Sweden. FAU - Sten, Liselotte AU - Sten L AD - Department of Pathology, Sahlgrenska University Hospital, Gothenburg, Sweden. FAU - Hashimi, Farida AU - Hashimi F AD - Department of Radiology, Sahlgrenska University Hospital, Gothenburg, Sweden. FAU - Svanvik, Joar AU - Svanvik J AD - Transplant Institute, Sahlgrenska University Hospital, Gothenburg, Sweden. FAU - Lindner, Per AU - Lindner P AD - Transplant Institute, Sahlgrenska University Hospital, Gothenburg, Sweden. LA - eng PT - Case Reports DEP - 20200430 PL - Switzerland TA - Case Rep Oncol JT - Case reports in oncology JID - 101517601 PMC - PMC7250374 OTO - NOTNLM OT - Checkpoint inhibition OT - Combined hepatocellular-cholangiocarcinoma OT - Immune-related hepatitis OT - Sorafenib COIS- The authors have no conflicts of interest to declare. EDAT- 2020/06/09 06:00 MHDA- 2020/06/09 06:01 PMCR- 2020/04/30 CRDT- 2020/06/09 06:00 PHST- 2020/03/16 00:00 [received] PHST- 2020/03/17 00:00 [accepted] PHST- 2020/06/09 06:00 [entrez] PHST- 2020/06/09 06:00 [pubmed] PHST- 2020/06/09 06:01 [medline] PHST- 2020/04/30 00:00 [pmc-release] AID - cro-0013-0478 [pii] AID - 10.1159/000507320 [doi] PST - epublish SO - Case Rep Oncol. 2020 Apr 30;13(1):478-484. doi: 10.1159/000507320. eCollection 2020 Jan-Apr.