An unusual malignant main bile duct stricture: a biliary metastasis of endometrial adenocarcinoma.

Biliary strictures are frequently encountered in interventional endoscopy. Their etiological diagnosis may be complicated [1]. Endoscopic ultrasound (EUS) guided biopsies, brush cytology, and wire-guided biopsies allow a diagnosis in most cases. Single-operator cholangioscopy (SOC) has radically changed the diagnostic approach, allowing visualization of the lesion, endoscopic characterization, and targeted biopsies [2].

We report the case of a 71-year-old woman with a history of endometrial adenocarcinoma, with muscular and bone recurrence 4 years after treatment. She developed a sudden jaundice without any other clinical signs. A magnetic resonance cholangiopancreatography found a circumferential parietal thickening of the main bile duct with dilatation of the intrahepatic bile ducts ([Fig. 1]). We decided to perform an EUS, which showed extensive cholangitis in the middle part of the bile duct with circumferential thickening of the bile duct mucosa. A 22 G needle biopsy was performed. Endoscopic retrograde cholangiopancreatography (ERCP) was then performed ([Fig. 2]) with SOC, which showed that the strictured area was indeed a fibrous stenosis with anarchic vascularization ([Video 1]).

Fig. 1 Biliary magnetic resonance cholangiopancreatography imaging (MRCP) showing the main bile duct stricture with dilatation of the intrahepatic bile ducts. a MRCP sequence showing the biliary stricture (red arrow) and biliary dilatation. b Three-dimensional reconstruction of the biliary system showing the biliary stenosis (red arrow).

Fig. 2 Retrograde cholangiography showing the different stages of the endoscopic retrograde cholangiopancreatography procedure. a Retrograde cholangiography showing the stricture of the main bile duct (black arrow). b Retrograde cholangiography with single-operator cholangioscopy (black arrow). c Retrograde cholangiography with wire-guided forceps (black arrow). d Placement of three plastic biliary stents.

Video 1 Endoscopic retrograde cholangiopancreatography and single-operator cholangioscopy for the diagnosis of indeterminate biliary stenosis and drainage with three plastic stents.

Biopsies were taken with forceps. Brush cytology and wire-guided biopsies were also performed. The procedure was completed with the placement of three plastic stents (one 15 cm and 8.5 Fr stent in the left bile duct; one 12 cm and 8.5 Fr stent and one 12 cm and 7 Fr stent in the right intrahepatic bile ducts). Histological examination found carcinomatous cells (mutated p53, PAX8 +), which were presumed to be metastasis of gynecological origin ([Fig. 3]).

Fig. 3 Biopsies of the main bile duct under single-operator cholangioscopy. a Normal biliary epithelium (black arrow) and neoplastic cells (red arrows) with hematoxylin-eosin stain (magnification × 200). b Neoplastic cells of gynecological origin with immunohistochemistry stain (PAX 8).

The use of SOC allows a finer analysis of indeterminate biliary stenosis. The presence of aberrant vascularization seems to be correlated with the neoplastic nature of the lesion [3]. SOC therefore allows macroscopic analysis of the lesion and targeted biopsies, probably making biliary sampling less random [4] [5].

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• References

• 1 Bowlus CL, Olson KA, Gershwin ME. Evaluation of indeterminate biliary strictures. Nat Rev Gastroenterol Hepatol 2016; 13: 28-37
• 2 Gerges C, Beyna T, Tang RSY. et al. Digital single-operator peroral cholangioscopy-guided biopsy sampling versus ERCP-guided brushing for indeterminate biliary strictures: a prospective, randomized, multicenter trial (with video). Gastrointest Endosc 2020; 91: 1105-1113
• 3 Robles-Medranda C, Oleas R, Sánchez-Carriel M. et al. Vascularity can distinguish neoplastic from non-neoplastic bile duct lesions during digital single-operator cholangioscopy. Gastrointest Endosc 2021; 93: 935-941
• 4 Angsuwatcharakon P, Kulpatcharapong S, Moon JH. et al. Consensus guidelines on the role of cholangioscopy to diagnose indeterminate biliary stricture. HPB (Oxford) 2021; 24: 17-19
• 5 Almadi MA, Itoi T, Moon JH. et al. Using single-operator cholangioscopy for endoscopic evaluation of indeterminate biliary strictures: results from a large multinational registry. Endoscopy 2020; 52: 574-582