Intraductal papillary neoplasm of bile duct with invasive carcinoma as an intrahepatic cystic lesion, with successful preoperative diagnosis

An 82-year-old man presented to the emergency department with abdominal pain and febrile symptoms that had been present for 4 days. Blood tests showed elevated liver enzymes and white blood cell count, and abdominal contrast-enhanced CT revealed a 35 mm cystic lesion in the left lateral liver lobe. On closer examination, the cystic lesion was found to have contiguous bile duct dilatation and internal nodules. Furthermore, mucus production was observed during endoscopic retrograde cholangiopancreatography, which led to the diagnosis of intraductal papillary neoplasm of the bile duct (IPNB), with cystic infection. Although the patient was an older adult, there was no background disease that would have prevented surgery, and resection was performed. Pathological examination revealed type 1 IPNB, with invasive carcinoma. The number of reports of IPNB is expected to increase with an increasing older population in Asia, and we report the findings of this case.

A case of hepatic intraductal papillary neoplasm of the bile duct

In this report, we present a 57-year-old female with a history of mild alcoholic liver disease during a medical check-up. Abdominal computed tomography and magnetic resonance imaging showed a multicystic mass with a solid enhancing mural nodule in the right lobe of the liver. Subsequently, laparoscopic right liver lobectomy was performed and pathological findings revealed intraductal papillary neoplasm of the bile duct (IPNB) with an associated invasive carcinoma. IPNB is a relatively rare disease that should be considered in the differential diagnosis of hepatic cystic tumours. Our case report highlights the importance of capturing image findings of the IPNB as this disease has a high potential for malignancy.

Recurrent intraductal papillary neoplasm of the bile duct due to intraductal dissemination: a case report and literature review

Background Intraductal papillary neoplasm of the bile duct (IPNB) is a subtype of biliary tumor. The 5-year survival rate of patients with IPNB who underwent curative resection is 81%. However, IPNB is known to often recur in other parts of the bile duct. Nevertheless, its mechanism remains poorly understood. Herein, we report the case of a patient with recurrent IPNB, which was considered to be attributed to intraductal dissemination in the common bile duct at 12 months after curative resection. We also made a review of the existing literature. Case presentation A 69-year-old man was referred to our hospital for the evaluation and dilation of an intrahepatic bile duct (IHBD) mass. Computed tomography (CT) findings confirmed a mass in the left hepatic duct. Left trisectionectomy, extrahepatic bile duct resection with biliary reconstruction, and regional lymph node dissection were performed. Intraoperative examination of the resection margin at the common bile duct and posterior segmental branch of the hepatic duct was negative for the presence of malignant cells. Histologically, the tumor showed intraductal papillary growth of the mucinous epithelium and was diagnosed as non-invasive IPNB. It had a papillary structure with atypical epithelial cells lined up along the neoplastic fibrovascular stalks. Immunohistochemically, this was as a gastric-type lesion. At 12 postoperative months, CT revealed a 1.5-cm mass in the lower remnant common bile duct. We performed subtotal stomach-preserving pancreaticoduodenectomy. The tumor exhibited papillary growth and was microscopically and immunohistochemically similar to the first tumor. At approximately 16 months after the patient’s second discharge, CT showed an abdominal mass at the superior mesenteric plexus, which was diagnosed as recurrent IPNB. Chemotherapy is ongoing, and the patient is still alive. In this case, as described in many previous reports, IPNB recurred below the primary lesion in the bile duct. Conclusion Based on our review of previous reports on IPNB recurrence, intraductal dissemination was considered one of the mechanisms underlying recurrence after multicentric development. Considering the high frequency and oncological conversion of recurrence in IPNB, regular follow-up examination is essential to achieve better prognosis in patients with recurrent IPNB.

Endoscopic Endoluminal Radiofrequency Ablation and Single-Operator Peroral Cholangioscopy System (SpyGlass) in the Diagnosis and Treatment of Intraductal Papillary Neoplasm of the Bile Duct: A Case Report and Literature Review

Background: Intraductal papillary neoplasm of the bile duct (IPNB) is a rare biliary benign tumor with atypical clinical features and is frequently misdiagnosed. Its treatment is limited and surgical resection is thought to be the only therapeutic option in patients with IPNB. With the aim of increasing the early diagnosis rate of IPNB and providing more therapeutic options for surgeons, we innovatively put forward the concept of combined utilization of SpyGlass and endoscopic endoluminal radiofrequency ablation (ERFA) in the diagnosis and treatment of IPNB.Case Presentation: An 85-year-old woman was referred to our hospital due to right upper quadrant abdominal pain. The image examinations indicated suspicious filling defects at the upper common bile duct. Further evaluation of SpyGlass cholangioscopy showed multiple reddish villous lesions at the left hepatic duct, and SpyBite biopsy under direct visualization demonstrated papillary low-grade dysplasia. In consideration of the advanced age and preference of the patient, the novel ERFA therapy was performed. The procedure was successful without periprocedural complications; the patient recovered uneventfully and was discharged 2 days after the operation. Upon follow-up, the patient was asymptomatic and in good physical condition at 8 months postoperatively.Conclusion: Preliminarily, we demonstrate that the strategy of a combination of SpyGlass and ERAF seems to be a promising, feasible, well-tolerated, and safe management for patients with IPNB. However, more data with larger patient volumes are needed to evaluate its outcomes further.