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©The Author(s) 2020. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. Jan 21, 2020; 26(3): 366-374
Published online Jan 21, 2020. doi: 10.3748/wjg.v26.i3.366
Unexpected metastasis of intraductal papillary neoplasm of the bile duct without an invasive component to the brain and lungs: A case report
Nguyen Hai Nam, Kojiro Taura, Masashi Kanai, Keita Fukuyama, Norimitsu Uza, Hirona Maeda, Yojiro Yutaka, Toyofumi F Chen-Yoshikawa, Manabu Muto, Shinji Uemoto
Nguyen Hai Nam, Kojiro Taura, Shinji Uemoto, Division of Hepato-Biliary-Pancreatic Surgery and Transplantation, Department of Surgery, Graduate School of Medicine, Kyoto University, Kyoto 606-8501, Japan
Masashi Kanai, Keita Fukuyama, Department of Clinical Oncology, Pharmacogenomics, and Palliative Medicine, Graduate School of Medicine, Kyoto University, Kyoto 606-8507, Japan
Norimitsu Uza, Department of Gastroenterology and Hepatology, Graduate School of Medicine, Kyoto University, Kyoto 606-8507, Japan
Hirona Maeda, Department of Diagnostic Pathology, Kyoto University Hospital, Kyoto 606-8507, Japan
Yojiro Yutaka, Toyofumi F Chen-Yoshikawa, Department of Thoracic. Surgery, Graduate School of Medicine, Kyoto University, Kyoto 606-8507, Japan
Manabu Muto, Department of Therapeutic Oncology, Graduate School of Medicine, Kyoto University, Kyoto 606-8507, Japan
Author contributions: Nam NH and Taura K designed this report, reviewed the literature and contributed to manuscript drafting; Taura K and Uemoto S were the patient’s HPB surgeons; Maeda H performed the pathological examination; Yutaka Y and Chen-Yoshikawa TF were the patient’s thoracic surgeons; Kanai M, Fukuyama K, Muto M performed the genomic profiling analysis using whole exome sequencing; Nam NH, Taura K and Uza N were responsible for the revision of the manuscript for important intellectual content; all authors issued final approval for the version to be submitted.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
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: Kojiro Taura, MD, PhD, Associate Professor, Doctor, Surgeon, Surgical Oncologist, Division of Hepato-Biliary-Pancreatic Surgery and Transplantation, Department of Surgery, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto 606-8507, Japan.
ktaura@kuhp.kyoto-u.ac.jp
Received: October 13, 2019
Peer-review started: October 13, 2019
First decision: December 5, 2019
Revised: December 13, 2019
Accepted: December 22, 2019
Article in press: December 22, 2019
Published online: January 21, 2020
Processing time: 94 Days and 18.4 Hours
BACKGROUND
Despite an expanding number of studies on intraductal papillary neoplasm of the bile duct (IPNB), distant metastasis remains unexplained especially in cases of carcinoma in situ. In the present study, we report a rare and interesting case of IPNB without invasive components that later metastasized to lungs and brain.
CASE SUMMARY
A 69-year-old male was referred to our hospital due to suspected cholangiocarcinoma. Laboratory tests on admission reported a mild elevation of alkaline phosphatase, γ-glutamyl transpeptidase, and total bilirubin in serum. Endoscopic retrograde cholangiography revealed a filling defect in the common bile duct (CBD) extending to the left hepatic duct. Peroral cholangioscopy delineated a tumor in the CBD that had a papillary pattern. Multidetector computed tomography and magnetic resonance cholangiopancreatography detected partial blockage ot interlude in the CBD leading to cholestasis without evidence of metastasis. Therefore, a diagnosis of IPNB cT1N0M0 was established. Left hepatectomy with bile duct reconstruction was performed. Pathological examination confirmed an intraepithelial neoplasia pattern without an invasive component and an R0 resection achievement. The patient was monitored carefully by regular examinations. However, at 32 mo after the operation, a 26 mm tumor in the lungs and a 12 mm lesion in the brain were detected following a suspicious elevated CA 19-9 level. Video-assisted thoracoscopic surgery of left upper lobectomy and stereotactic radiotherapy are indicated. In addition to histopathological results, a genomic profiling analysis using whole exome sequencing subsequently confirmed lung metastasis originating from bile duct cancer.
CONCLUSION
This case highlights the important role of genomic profiling analysis using whole exome sequencing in identifying the origin of metastasis in patients with IPNB.
Core tip: An intraductal papillary neoplasm of the bile duct without an invasive component has unexpectedly metastasized to the brain and lungs. In addition to histopathological results, a genomic profiling analysis using whole exome sequencing subsequently confirmed lung metastasis originating from bile duct cancer by detecting 100 single nucleotide variants and 168 insertion/deletions.
