Intraductal Papillary Neoplasm of the Bile Duct in a Patient with Recurrent Obstructive Jaundice Postcholecystectomy: A Balloon Sweep of Serendipity!

Dear Editor, We read with keen interest a recently published case report “Intraductal papillary neoplasm of the bile duct (IPNB)” with a unique clinical presentation of “recurrent acute pancreatitis.”[1] The authors have elaborated in detail regarding the management of the patient, along with a brief discussion of this relatively uncommon preneoplastic lesion of the bile duct. Step-wise evaluation in the form of (i) altered liver function test parameters, (ii) magnetic resonance imaging (MRI) demonstrating an ill-defined lesion in the right hepatic duct with upstream biliary radicle dilatation, and (iii) cholangioscopy-guided biopsy of the frond-like intraductal papillary lesion led to the diagnosis of IPNB (gastric subtype with low-grade dysplasia) involving the right hepatic duct. However, what remains intriguing and rather unanswered is the “pathogenesis” of pancreatitis episodes in this given patient. We wish to highlight our recent experience in the management of another interesting clinical presentation of this rare preneoplastic lesion of the bile duct in an elderly gentleman. A 75-year-old gentleman presented with obstructive jaundice/cholangitis (fever, icterus, right upper quadrant pain, elevated total/direct bilirubin, alkaline phosphatase, and MRI/Magnetic resonance cholangiopancreaticography (MRCP) demonstrating cholelithiasis and filling defects in the proximal common bile duct (CBD) suspicious of calculi and sludge. After confirmation of these findings on an endoscopic ultrasound (EUS) examination, endoscopic retrograde cholangiography (ERCP), CBD clearance, and biliary plastic stent insertion were performed. Balloon sweeps revealed few calculi and sludge. The patient later underwent routine laparoscopic cholecystectomy followed by side-view endoscopy-guided CBD stent removal elsewhere. Six months later, he again presented with features suggestive of obstructive jaundice and cholangitis. The MRCP done elsewhere was reported as dilated, thick-wall proximal CBD with intraluminal filling defects raising suspicion for residual/recurrent choledocholithiasis. We went ahead with ERCP and ductal clearance. To our surprise, balloon sweeps revealed a tissue-like material mixed with blood clots and copious amount of sludge which adhered to the tip of the endoscope on applying suction. The scope was gradually withdrawn, and the adherent tissue-like material was collected in a formalin container and sent for histopathology examination [Figure 1]. Subsequent occlusion cholangiogram showed a persistent, irregular filling defect in the CHD extending beyond the hilum into the right hepatic duct despite multiple balloon sweeps [Figure 2]. In view of the uncertain nature of the intraductal lesion at the hilum and the presence of cholangitis, bilateral 10 Fr. plastic stent insertion was performed. Logistic reasons precluded a cholangioscopy examination in the same setting. An EUS examination done post-ERCP biliary stenting revealed an irregular intraductal hypoechoic lesion in the dilated (11.5 mm) proximal CBD without any adjacent periportal lymph nodes or peri-ductal mass lesion [Figure 3]. Histopathology of the serendipitously acquired tissue-sample was reported as IPNB of the bile duct (pancreatico-biliary type with high-grade dysplasia) [Figure 4]. The patient was offered the option of upfront surgery versus localized ablative therapy (intraductal radiofrequency ablation followed by bilateral uncovered self-expanding metal stent placement). He underwent extended bile duct resection and hepaticojejunostomy after confirmation of localized nature of the lesion on positron emission tomography computed tomography. He has been asymptomatic till last follow-up recorded at 6 months postsurgery.Figure 1: Tissue-like material mixed with blood clots on balloon sweep (a) which was retrieved and sent for sent for histopathology in a formalin container (b)Figure 2: Occlusion cholangiogram demonstrating persistent intra-ductal filling defect at the hilum after multiple balloon sweepsFigure 3: Endoscopic ultrasound demonstrating an irregular, hypoechoic lesion in the dilated proximal common bile ductFigure 4: Histopathology pictures of hematoxylin and eosin stain showing both low (a) and high-power (b) images of a papillary neoplasm comprising of complex tubulo-villous and micropapillary pattern with mitotic activity s/o pancreatico-biliary type of intraductal papillary neoplasm of the bile duct with high-grade dysplasiaThis case demonstrates an incidental diagnosis of IPNB of the bile duct in a patient with recurrent jaundice and suspected residual/recurrent choledocholithiasis postcholecystectomy. Although our patient did not present with acute pancreatitis, we are of the opinion that IPNB of the bile duct can potentially present with acute pancreatitis (such as the previously published case) due to hypothetical risk of obstruction of the common channel/main pancreatic duct due to sludge/calculi resulting from biliary stasis or from mucin hypersecretion and extrusion of papillary fronds from the intraductal tumor.[2] Declaration of patient consent The authors certify that they have obtained all appropriate patient consent forms. In the form, the legal guardian has given his consent for images and other clinical information to be reported in the journal. The guardian understands that names and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed. Financial support and sponsorship Nil. Conflicts of interest There are no conflicts of interest.