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GE-Portuguese Journal of Gastroenterology

versão impressa ISSN 2341-4545

GE Port J Gastroenterol vol.29 no.3 Lisboa jun. 2022  Epub 02-Jan-2023

https://doi.org/10.1159/000516007 

Endoscopic Snapshot

Recurrent Acute Pancreatitis with a Bulging Papilla: An Unexpected Diagnosis

Pancreatite aguda recorrente em doente com papila tumefacta: um diagnóstico inesperado

Marta Moreira1 
http://orcid.org/0000-0002-3907-3165

João Fernandes1 
http://orcid.org/0000-0002-3759-2383

Tarcísio Araújo1 

Richard Azevedo1 

Jorge Canena2  3 
http://orcid.org/0000-0002-2539-5876

Luis Lopes1  4  5 
http://orcid.org/0000-0002-8835-0923

1Department of Gastroenterology, Santa Luzia Hospital, Unidade Local de Saúde Alto Minho, Viana do Castelo, Portugal;

2Department of Gastroenterology, Professor Doutor Fernando Fonseca Hospital, Amadora, Portugal;

3Department of Gastroenterology, Nova Medical School/Faculty of Medical Sciences, Lisbon, Portugal

4Life and Health Sciences Research Institute (ICVS), School of Medicine, University of Minho, Braga, Portugal;

5ICVS/3B’s, PT Government Associate Laboratory, Braga/Guimarães, Portugal


Bile duct adenomas are a very rare entity, mostly located in the gallbladder and incidentally found in surgical specimens; however, they can be detected in any segment of the biliary tree [1]. The natural history of these lesions is not well established but is postulated to be similar to the adenoma-adenocarcinoma sequence existing elsewhere in the gastrointestinal tract [2].

We present the case of an 87-year-old man with a history of two episodes of acute pancreatitis, with a 3-month interval between them, referred for upper endoscopic ultrasound. This study showed a 15 × 10 mm isoechoic image in the terminal portion of the common bile duct (CBD), with a heterogeneous aspect and no acoustic shadowing (Fig. 1). A subsequent endoscopic retrograde cholangiopancreatography (ERCP) was performed, revealing an enlarged and bulging papilla, with normal overlying mucosa (Fig. 2a). The cholangiogram revealed a filling defect in the distal third of the CBD, approximately 20 mm in diameter, compatible with a bile duct stone (Fig. 2b). After biliary sphincterotomy, a biliary sweepage was performed using a Dormia basket revealing a villous polypoid lesion of about 20 mm extruding from the bile duct into the duodenum (Fig. 2c). Biopsies were taken and confirmed it to be a villous adenoma with low-grade dysplasia (Fig. 3). Given the patient’s age and comorbidities, surgery was precluded, and resection was performed using a hot snare polypectomy technique, followed by ablation with argon plasma. The etiology of the acute pancreatitis was attributed to the adenoma, after exclusion of more common etiologies (gallstones, alcohol consumption, potential drugs or toxins, triglycerides, hypercalcemia, IgG4 and autoimmune pancreatitis or anatomic anomalies). Given the patient’s age and no relevant family history, genetic etiologies were not investigated.

Fig. 1 Echoendoscopic image showing a 15 × 10 mm isoechoic image in the distal CBD with a heterogeneous aspect without shadow cone. 

Fig. 2 ERCP images. a Endoscopic view showing a bulging papilla with normal overlying mucosa. b 20-mm filling defect in the distal CBD compatible with a biliary stone. c Biliary sweepage with a Dormia basket revealed a 20-mm villous polypoid lesion emanating from the bile duct into the duodenum. 

Fig. 3 Biopsy histology showing epithelium-coated papillae without atypia (HE, ×40). 

The patient was lost to follow-up during a 4-year period and was again referred to the Gastroenterology Department because of new-onset jaundice. An ERCP was performed showing adenomatous tissue in the terminal portion of the CBD, and biopsies were compatible with adenoma with low-grade dysplasia. Given the patient’s contraindications for a surgical approach, bipolar radiofrequency (HabibTM) of the adenomatous tissue was performed with technical success. A follow-up ERCP with cholangioscopy is already scheduled.

Adenomas arising from the extrahepatic biliary tree are still poorly understood, mainly because of its very low incidence. Nevertheless, current knowledge supports the belief that these lesions may potentially evolve into carcinomas, like adenomas located elsewhere [1]. Most authors believe that removal of these adenomatous lesions may prevent the development of adenocarcinoma [1-3]. Little is known about the use of radiofrequency in biliary adenomas. In the few published case reports, a surgical approach was used. A minority of patients was maintained in surveillance without invasive treatment due to patients’ risks and poor life expectancy [1].

We highlight this case because of the rarity of this pathology, the presentation as recurrent acute pancreatitis that, to our knowledge, is not described in the small number of case series published [1], and because of the diagnostic challenge it posed.

Consult the ERCP endoscopic video available at the following link: 0

References

1. Loh KP, Nautsch D, Mueller J, Desilets D, Mehendiratta V. Adenomas involving the extrahepatic biliary tree are rare but have an aggressive clinical course. Endosc Int Open. 2016 Feb;4(2):E112-7. [ Links ]

2. Čekas K, Rudaitis V, Beiša V, Jotautas V, Rutkauskaitė D, Meškauskas R, et al. Common bile duct villous adenoma: a case report and review of the literature. J Med Case Reports. 2016 Jan;10(1):18. [ Links ]

3. Hacking CP, Taylor CJ, Nathanson LK. Common bile duct polyp mimicking choledocholithiasis: a case report with laparoscopic transcystic management. Surg Laparosc Endosc Percutan Tech. 2008 Jun;18(3):290-3. [ Links ]

1Statement of Ethics The subject has given his written informed consent to publish the case (including publication of images).

Funding Sources This study did not require any funding.

Received: November 25, 2020; Accepted: March 06, 2021

Corresponding author Marta Moreira marta.isabel.moreira@ulsam.min-saude.pt

Conflict of Interest Statement The authors have no conflicts of interest to declare.

Author Contributions M.M., J.F., and L.L. wrote the manuscript. R.A., J.C., and T.A. were responsible for the revision of its contents

Creative Commons License This is an open-access article distributed under the terms of the Creative Commons Attribution License