Nacif LS, Hessheimer AJ, Rodríguez Gómez S, Montironi C, Fondevila C. Infiltrative xanthogranulomatous cholecystitis mimicking aggressive gallbladder carcinoma: A diagnostic and therapeutic dilemma. World J Gastroenterol 2017; 23(48): 8671-8678 [PMID: 29358875 DOI: 10.3748/wjg.v23.i48.8671]
Corresponding Author of This Article
Constantino Fondevila, MD, PhD, Associate Professor of Surgery, Hepatobiliary Surgery and Liver Transplant, Hospital Clínic, University of Barcelona, C/ Villarroel 170, Barcelona 08036, Spain. cfonde@clinic.ub.es
Research Domain of This Article
Surgery
Article-Type of This Article
Case Report
Open-Access Policy of This Article
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/
Lucas Souto Nacif, Amelia Judith Hessheimer, Constantino Fondevila, Department of Surgery, Institut de Malalties Digestives I Metabòliques (IMDM), Hospital Clínic, Barcelona 08036, Spain
Lucas Souto Nacif, Amelia Judith Hessheimer, Constantino Fondevila, CIBERehd, IDIBAPS, University of Barcelona, Barcelona 08036, Spain
Sonia Rodríguez Gómez, Department of Radiology, Hospital Clínic, Barcelona 08036, Spain
Carla Montironi, Department of Pathology, Hospital Clínic, Barcelona 08036, Spain
Author contributions: Nacif LS and Hessheimer AJ contributed equally to this work; Nacif LS and Fondevila C designed the work; Nacif LS, Hessheimer AJ, Rodríguez Gómez S and Montironi C acquired and analyzed the data; Nacif LS and Hessheimer AJ wrote the manuscript; Rodríguez Gómez S, Montironi C and Fondevila C provided critical appraisal of the manuscript.
Supported by Nacif LS was supported by an International Travel Scholar Award from the International Liver Transplantation Society (ILTS).
Informed consent statement: All study participants provided informed consent prior to study enrollment.
Conflict-of-interest statement: None of the authors have any conflicts of interest to declare.
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/
Correspondence to: Constantino Fondevila, MD, PhD, Associate Professor of Surgery, Hepatobiliary Surgery and Liver Transplant, Hospital Clínic, University of Barcelona, C/ Villarroel 170, Barcelona 08036, Spain. cfonde@clinic.ub.es
Telephone: +34-93-2275718 Fax: +34-93-2275589
Received: September 13, 2017 Peer-review started: September 13, 2017 First decision: October 10, 2017 Revised: October 13, 2017 Accepted: November 21, 2017 Article in press: November 21, 2017 Published online: December 28, 2017 Processing time: 105 Days and 3.2 Hours
ARTICLE HIGHLIGHTS
Case characteristics
Three patients presented with jaundice and variable other symptoms, including abdominal pain and weight loss.
Clinical diagnosis
Clinical findings were suggestive of neoplastic processes affecting directly or indirectly the biliary tree.
Differential diagnosis
Serum bilirubin was elevated in all three cases, while serum CA-19.9 levels were normal.
Laboratory diagnosis
Laboratory tests and imaging studies were performed to clarify the diagnosis.
Imaging diagnosis
Abdominal imaging studies, including CT and magnetic resonance cholangiopancreatography, demonstrated widely infiltrative hilar mass lesions with associated vascular affectation and retrograde biliary dilatation.
Pathological diagnosis
Since all three patients had aggressive yet apparently resectable lesions, surgery was undertaken without previous biopsy.
Treatment
All three interventions were performed according to oncological principles and included, at a minimum, radical cholecystectomy, common bile duct excision, hilar lymphadenectomy, and hepaticojejunostomy.
Related reports
There are a few previous reports that describe radical resection of very aggressive cases of what ultimately turned out to be xanthogranulomatous cholecystitis, and most describe little-to-no postoperative morbidity or mortality.
Term explanation
In xanthogranulomatous cholecystitis, mucin and bile are extravasated into subepithelial tissues and phagocytosed, resulting in inflammation, xanthoma formation, and processes of repair and fibrosis that, in some cases, produce pseudotumors that may be confused with malignancy.
Experiences and lessons
For clinicians confronting similar cases, we recommend direct surgical intervention performed by an experienced hepatobiliary surgeon capable of removing all diseased tissue, reconstructing the patient’s anatomy, and effectively curing the patient of his or her disease process.
