Case Report
Copyright ©The Author(s) 2019. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Aug 26, 2019; 7(16): 2302-2308
Published online Aug 26, 2019. doi: 10.12998/wjcc.v7.i16.2302
Primary malignant melanoma of the biliary tract: A case report and literature review
Soledad Cameselle-García, José Luis Fírvida Pérez, María C Areses, Jesús Daniel Fernández de Castro, Juan Mosquera-Reboredo, Jesús García-Mata
Soledad Cameselle-García, José Luis Fírvida Pérez, María C Areses, Jesús García-Mata, Department of Medical Oncology, Complejo Hospitalario Universitario de Ourense, SERGAS, Ourense 32005, Spain
Jesús Daniel Fernández de Castro, Department of Digestive, Complejo Hospitalario Universitario de Ourense, SERGAS, Ourense 32005, Spain
Juan Mosquera-Reboredo, Department of Pathology, Hospital San Rafael, A Coruña 15009, Spain
Author contributions: Cameselle-García S collected the patient’s clinical data and wrote the paper; Fírvida Pérez JL and Areses MC designed the report; García-Mata J revised the manuscript; Fernández de Castro JD reviewed the relevant literature and contributed to manuscript drafting; Mosquera-Reboredo JM performed the anatomopathological and histopathological analysis and contributed to manuscript drafting; All authors issued final approval for the version to be submitted.
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 (2013), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (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: Soledad Cameselle-García, MD, Medical Oncologist, Department of Medical Oncology, Complejo Hospitalario Universitario de Ourense, C/ Ramón Puga Noguerol, 54, Ourense 32005, Spain. soledad.cameselle.garcia@sergas.es
Telephone: +34-605534509 Fax: +34-988385551
Received: March 25, 2019
Peer-review started: March 25, 2019
First decision: June 4, 2019
Revised: June 29, 2019
Accepted: July 27, 2019
Article in press: July 27, 2019
Published online: August 26, 2019
Processing time: 158 Days and 4 Hours
Abstract
BACKGROUND

Primary malignant melanoma of the biliary tract (MBT) is a rare condition whose diagnosis requires excluding a primary origin in another location. This paper reviews the most important characteristics of MBT cases published in the literature and reports a new case. The patient reported here is the first case of primary malignant melanoma of the biliary tract with pulmonary metastasis treated with immunotherapy. This patient remains disease-free 36 mo after the treatment of metastatic lung lesions.

CASE SUMMARY

A 51-year-old man was admitted to the gastrointestinal department to study obstructive jaundice of a 1 wk clinical course. Magnetic resonance cholangiopancreatography revealed dilatation of the intrahepatic biliary tract and stenosis of the common hepatic duct. Given the suspicion of biliary tract neoplasia, cholecystectomy and resection of the common hepatic duct were performed with hepatic jejunostomy free of complications. Anatomo-pathological diagnosis was melanoma. After intervention, the patient was referred to the Department of Medical Oncology, where a primary origin was excluded in the skin, mucosa, and eyes. This confirmed diagnosis of primary biliary tract melanoma. Computed tomography was performed 12 mo after the procedure revealed several subcentimetric lung nodules. Wedge resection was performed. After confirming the diagnosis of pulmonary metastasis of primary melanoma of the biliary tract, the patient was started on immunotherapy with nivolumab. Tolerance to treatment was excellent. The patient remains disease-free 36 mo after the treatment of metastatic lung lesions.

CONCLUSION

The patient reported here is the first case of primary malignant melanoma of the biliary tract with lung metastases successfully treated with immunotherapy.

Keywords: Biliary tract; Case report; Immunotherapy; Malignant melanoma

Core tip: Primary malignant melanoma of the biliary tract is a very rare entity that mainly affects men (men/women ratio 12:2) and has an average age of presentation of 47 years (range: 26-67). Cases usually present as abdominal pain and jaundice. Lesions are usually black, polypoid, and have endoluminal growth. In cases of localised disease, the treatment of choice is surgery. Mortality because of this disease was 50% after average follow-up of 37 mo (range: 4-204 mo). Here, for the first time, we describe a case of primary malignant melanoma of the biliary tract with pulmonary metastases, successfully treated with immunotherapy.