Ren Xu, Zhu Chun-Lan, Qin Xu-Fu, Jiang Hong, Xia Tian, Qu Yong-Ping
Digestive Disease Hospital of Heilongjiang Provincial Hospital, Harbin 150001, Heilongjiang Province, China.
The Second Digestive Department, Digestive Disease Hospital of Heilongjiang Provincial Hospital, Harbin 150001, Heilongjiang Provice, China.
World J Clin Cases. 2019 Jan 6;7(1):102-108. doi: 10.12998/wjcc.v7.i1.102.
Intraductal papillary neoplasm of the bile duct (IPNB) is pathologically similar to intraductal papillary mucinous neoplasm (IPMN). However, there are several significant differences between them. The rate of IPMN associated with extrapancreatic malignancies has been reported to range from 10%-40%, and it may occasionally be complicated with the presence of fistulas. IPMN associated with malignant IPNB is extremely rare and only nine cases have been reported in the literature.
We report a 52-year-old man who presented with recurrent cholangitis for nine months. Computed tomography and magnetic resonance cholangiopancreatography showed the common bile duct stricture with dilated pancreatobiliary duct without other abnormal findings. The underlying pathogenesis could not be identified based on the radiologic images. Endoscopic retrograde cholangiopancreatography revealed a pancreatobiliary fistula with dilated main pancreatic duct, biliary stricture with dilated biliary tree, and mucus discharge from the enlarged orifice of the major papilla. The patient underwent SpyGlass cholangiopancreatoscopy due to a suspected mucin-producing biliary neoplasm and indeterminate main pancreatic duct dilatation. Multiple papillary growing neoplasms with vascular images, with the extent of lesions spreading in the biliopancreatic ductal lumens, were identified by SpyGlass. In addition, the presence of a pancreatobiliary fistula was also identified. The patient was diagnosed as having benign IPMN and malignant IPNB with focal invasion by postoperative pathology. Furthermore, varying histological subtypes were present in both IPMN and IPNB. Pylorus-preserving pancreaticoduodenectomy was performed on the patient with excellent results during the 52 month follow-up period.
We deemed that pancreatography and SpyGlass allowed for an efficient diagnosis of IPMN with pancreatobiliary fistula, whereas the etiology could not be identified by radiologic imaging.
胆管内乳头状肿瘤（IPNB）在病理上与导管内乳头状黏液性肿瘤（IPMN）相似。然而，它们之间存在一些显著差异。据报道，与胰腺外恶性肿瘤相关的IPMN发生率为10%-40%，偶尔可能并发瘘管。与恶性IPNB相关的IPMN极为罕见，文献中仅报道了9例。
我们报告一名52岁男性，因反复胆管炎9个月就诊。计算机断层扫描和磁共振胰胆管造影显示胆总管狭窄伴胰胆管扩张，无其他异常发现。根据影像学图像无法确定潜在的发病机制。内镜逆行胰胆管造影显示胰胆管瘘伴主胰管扩张、胆管狭窄伴胆管树扩张，以及主乳头扩大开口处有黏液排出。由于怀疑存在产生黏液的胆管肿瘤和主胰管扩张不明确，该患者接受了SpyGlass胰胆管镜检查。SpyGlass检查发现多个有血管影像的乳头状生长肿瘤，病变范围在胆胰管腔内扩散。此外，还发现了胰胆管瘘的存在。术后病理诊断为良性IPMN和恶性IPNB伴局灶性侵犯。此外，IPMN和IPNB中均存在不同的组织学亚型。对该患者实施了保留幽门的胰十二指肠切除术，在52个月的随访期内效果良好。
我们认为胰胆管造影和SpyGlass能够有效诊断伴有胰胆管瘘的IPMN，而影像学检查无法确定病因。
