Ampullectomy of an unusual lesion developing 20 years after endoscopic treatment of a type III choledochocele

A type III choledochocele is a rare congenital cystic dilation of the intraduodenal portion of the main bile duct, representing 5 % of all choledochal cysts according to the Toldani classification [1]. Choledochoceles may cause biliary obstruction and carcinoma development with a reported incidence of 2.5 %. To date, endoscopic treatment remains the less invasive technique, which consists of cystic mass removal associated with sphincterotomy [2] [3]. However, the risk of carcinoma development may persist even after treatment and reports of long-term outcomes are lacking [4].

Herein we report the original case of a 78-year-old woman presenting with a previous history of sphincterotomy for a type III choledochocele 20 years ago ([Video 1]). She presented with a 12-mm low-grade dysplasia adenoma arising from the sphincterotomy scar ([Fig. 1]), mimicking an ampulloma. To the best of our knowledge, such a lesion has yet to be reported. Imaging modalities (endoscopic ultrasound and magnetic resonance imaging) revealed a 9-mm moderate dilation of the main bile duct upstream of a distal stenosis ([Fig. 2]).

Video 1 Ampullectomy of an unusual lesion that developed 20 years after endoscopic treatment of a type III choledochocele.

Fig. 1 Endoscopic view of the ampulloma-like lesion involving the biliary tract and the whole ampulloma mass.

Fig. 2 Endoscopic aspect of the duodenal wall after resection.

A new endoscopic procedure was performed. The patient was placed in a dorsal position under general anesthesia. Similar to an ampullectomy, a duodenoscope under CO2 insufflation was used. An en bloc resection was performed using a 15-mm braided snare (Lariat; Life Partners Europe, Bagnolet, France) and the specimen was immediately retrieved ( [Fig.3]). Cholangiography revealed a delayed clearance of the contrast due to the distal stenosis ( [Fig.4]). Complementary intraductal biopsies of the stenosis were performed using a pediatric biopsy forceps. Prevention of post-ampullectomy pancreatitis was ensured by intrarectal indometacine administration, pancreatic stenting with a 5-Fr × 5-cm plastic stent, and ringer lactate hyperhydration. The patient was discharged after 24 h without complications.

Fig. 3 Additional biopsies of the stenosis were guided by cholangiography. Cholangiography revealed a distal stenosis of the main bile duct. Biopsy sampling of the stenosis was performed using pediatric biopsy forceps.

Fig. 4 Histological features of the ampulloma-like resection. Hematoxylin and eosin staining revealed a tubulous adenoma with low-grade dysplasia arising and surrounding the biliary orifice.

Final histology confirmed the R0 resection of a low-grade dysplasia adenoma. Biopsies of the stenosis revealed only fibrotic tissue. The 4-month follow-up duodenoscopy confirmed the absence of local recurrence.

Endoscopy_UCTN_Code_CCL_1AZ

Competing interests

The authors declare that they have no conflict of interest.

• References

• 1 Todani T, Watanabe Y, Narusue M. et al. Congenital bile duct cysts: classification, operative procedures, and review of thirty-seven cases including cancer arising from choledochal cyst. Am J Surg 1977; 134: 263-269
• 2 Culetto A, Miranda-Garcia P, Tellechea JI. et al. Endoscopic treatment of a choledochocele. Endoscopy 2016; 48: E286
• 3 Lobeck IN, Dupree P, Falcone RA. et al. The presentation and management of choledochocele (type III choledochal cyst): a 40-year systematic review of the literature. J Pediatr Surg 2017; 52: 644-649
• 4 Kim TH, Park JS, Lee SS. et al. Carcinoma arising in choledochocele: is choledochocele innocent bystander or culprit?. Endoscopy 2002; 34: 675-676

Publication History

Article published online:
05 March 2021