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Rare variant of pancreaticobiliary maljunction associated with pancreas divisum in a child diagnosed and treated by endoscopic retrograde cholangiopancreatography: A case report

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World Journal of Clinical Cases 2019年第9期7卷 1073-1079页

作者： Guang-Xing Cui Hai-Tao Huang Jian-Feng Yang Xiao-Feng Zhang Department of Gastroenterology Affiliated Hangzhou First People’s Hospital Zhejiang University School of Medicine

BACKGROUND pancreaticobiliary maljunction(PBM) is an uncommon congenital anomaly of the pancreatic and biliary ductal system, defined as a union of the pancreatic and biliary ducts located outside the duodenal wall. According to the Komi classification of PBM, the common bile duct(CBD) directly fuses with the ventral pancreatic duct in all types. Pancreas divisum(PD) occurs when the ventral and dorsal ducts of the embryonic pancreas fail to fuse during the second month of fetal development. The coexistence of PBM and PD is an infrequent ***, we report an unusual variant of PBM associated with PD in a pediatric patient, in whom an anomalous communication existed between the CBD and dorsal pancreatic *** SUMMARY A boy aged 4 years and 2 mo was hospitalized for abdominal pain with nausea and jaundice for 5 d. Abdominal ultrasound showed cholecystitis with cholestasis in the gallbladder, dilated middle-upper CBD, and a strong echo in the lower CBD, indicating biliary stones. The diagnosis was extrahepatic biliary obstruction caused by biliary stones, which is an indication for endoscopic retrograde cholangiopancreatography(ERCP). ERCP was performed to remove biliary stones. During the ERCP, we found a rare communication between the CBD and dorsal pancreatic duct. After clearing the CBD with a balloon, an 8.5 Fr 4-cm pigtail plastic pancreatic stent was placed in the biliary duct through the major papilla. Six months later, his biliary stent was removed after he had no symptoms and normal laboratory tests. In the following 4-year period, the child grew up normally with no more attacks of abdominal *** We consider that ERCP is effective and safe in pediatric patients with PBM combined with PD, and can be the initial therapy to manage such cases,especially when it is combined with aberrant communication between the CBD and dorsal pancreatic duct.

关键词： pancreaticobiliary maljunction Pancreas divisum Endoscopic retrograde cholangiopancreatography Variant Communication Children Case report

Endoscopic retrograde cholangiopancreatography in children with symptomatic pancreaticobiliary maljunction: A retrospective multicenter study

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World Journal of Gastroenterology 2019年第40期25卷 6107-6115页

作者： Jing-Qing Zeng Zhao-Hui Deng Kai-Hua Yang Tian-Ao Zhang Wen-Yu Wang Jian-Mei Ji Ya-Bin Hu Chun-Di Xu Biao Gong Department of Pediatric Digestive Diseases Shanghai Children’s Medical CenterShanghai Jiao Tong University School of MedicineShanghai 200127China Department of Digestive Diseases Shanghai Shuguang HospitalShanghai University of Traditional Chinese MedicineShanghai 201203China Department of Clinical Epidemiology and Biostatistics Shanghai Children’s Medical CenterShanghai Jiao Tong University School of MedicineShanghai 200127China Department of Pediatrics Ruijin HospitalShanghai Jiao Tong University School of MedicineShanghai 200025China

BACKGROUND Endoscopic retrograde cholangiopancreatography(ERCP)has been widely used in pediatric patients with cholangiopancreatic *** To evaluate the efficacy,safety,and long-term follow-up results of ERCP in symptomatic pancreaticobiliary maljunction(PBM).METHODS A multicenter,retrospective study was conducted on 75 pediatric patients who were diagnosed with PBM and underwent therapeutic ERCP at three endoscopy centers between January 2008 and March *** were divided into four PBM groups based on the fluoroscopy in *** clinical characteristics,specific ERCP procedures,adverse events,and long-term follow-up results were retrospectively *** Totally,112 ERCPs were performed on the 75 children with symptomatic *** manifestations included abdominal pain(62/75,82.7%),vomiting(35/75,46.7%),acholic stool(4/75,5.3%),fever(3/75,4.0%),acute pancreatitis(47/75,62.7%),hyperbilirubinemia(13/75,17.3%),and elevated liver enzymes(22/75,29.3%).ERCP interventions included endoscopic sphincterotomy,endoscopic retrograde biliary or pancreatic drainage,stone extraction,***-related complications were observed in 12 patients and included post-ERCP pancreatitis(9/75,12.0%),gastrointestinal bleeding(1/75,1.3%),and infection(2/75,2.7%).During a mean follow-up period of 46 mo(range:2 to 134 mo),ERCP therapy alleviated the biliary obstruction and reduced the incidence of *** overall effective rate of ERCP therapy was 82.4%;seven patients(9.3%)were lost to follow-up,eight(11.8%)re-experienced pancreatitis,and eleven(16.2%)underwent radical surgery,known as prophylactic excision of the extrahepatic bile duct and *** ERCP is a safe and effective treatment option to relieve biliary or pancreatic obstruction in symptomatic PBM,with the characteristics of minor trauma,fewer complications,and repeatability.

关键词： Cholangiopancreatic diseases Endoscopic retrograde cholangiopancreatography pancreaticobiliary maljunction Pancreatitis Pediatric patients Follow-up

Carcinogenesis and chemoprevention of biliary tract cancer in pancreaticobiliary maljunction

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World Journal of Gastrointestinal Oncology 2010年第3期2卷 130-135页

作者： Akihiko Tsuchida Takao Itoi the third Department of Surgery Tokyo Medical University the fourth Department of Internal Medicine Tokyo Medical University

pancreaticobiliary maljunction (PBM) is a high risk factor for biliary tract cancer. In PBM, since the pancreatic duct and bile duct converge outside the duodenal wall beyond the influence of the sphincter of Oddi, pancreatic juice and bile are constantly mixed, producing a variety of harmful substances. Because of this, the biliary mucosa is repeatedly damaged and repaired, which causes an acceleration of cell proliferative activity and multiple gene mutations. Histological changes such as hyperplasia, metaplasia, and dysplasia ultimately result in a high incidence of carcinogenesis. In a nationwide survey by the Japanese Study Group on PBM, coexisting biliary tract cancer was detected in 278 of the 1627 registered cases of PBM (17.1%). Of these cases, in those with dilatation of the extrahepatic bile duct, cancer was often detected not only in the gallbladder but also in the bile ducts. More than 90% of cancer cases without dilatation of the extrahepatic bile duct develop in the gallbladder. Standard treatment for PBM is a cholecystectomy and resection of the extrahepatic bile duct. However, cholecystectomy alone is performed at nearly half of institutions in Japan. Conversely, reports of carcinogenesis in the remnant bile duct or pancreas after diversion surgery are steadily increasing. One of the causes for this is believed to be an accumulation of gene mutations which were present before surgery. Anticancer drugs are ineffective in preventing such carcinogenesis following surgery, thus the postoperative administration of chemopreventive agents may be necessary.

关键词： Chemoprevention Gallbladder cancer Bile duct cancer Carcinogenesis pancreaticobiliary maljunction

pancreaticobiliary reflux as a high-risk factor for biliary malignancy:Clinical features and diagnostic advancements

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World Journal of Hepatology 2015年第13期7卷 1735-1741页

作者： Reiji Sugita Department of Radiology Sendai City Medical Center

pancreaticobiliary junction is composed of complex structure with which biliary duct and pancreatic duct assemble and go out into the ampulla of Vater during duodenum wall surrounding the sphincter of *** the sphincter of Oddi functionally prevents the reflux of pancreatic juice,pancreaticobiliary reflux(PBR) occurs when function of the sphincter of Oddi *** anatomically abnormal junction is termed pancreaticobiliary maljunction(PBM) and is characterized by pancreatic and bile ducts joining outside of the duodenal *** is an important anatomical finding because many studies have revealed that biliary malignancies are related due to the carcinogenetic effect of the pancreatic back flow on the biliary *** the other hand,several studies have been published on the reflux of pancreatic juice into the bile duct without morphological PBM,and the correlation of such cases with biliary diseases,especially biliary malignancies,is drawing considerable *** it has long been possible to diagnose PBM by various imaging modalities,PBR without PBM has remained difficult to ***,the pathological features of PBR without PBM have not been yet fully ***,a new method of diagnosing PBR without PBM has appeared,and the features of PBR without PBM should soon be better understood.

关键词： pancreaticobiliary maljunction Pancreasjuice Reflux Flow Magnetic resonance imaging

Endoscopic diagnosis of pancreaticobiliary maljunction

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World Journal of Gastrointestinal Endoscopy 2011年第1期3卷 1-5页

作者： Terumi Kamisawa Kensuke Takuma Fumihide Itokawa Takao Itoi Department of Internal Medicine Tokyo Metropolitan Komagome Hospital Department of Gastroenterology Tokyo Medical University

pancreaticobiliary maljunction (PBM) is a congenital anomaly defined as a junction of the pancreatic and bile ducts located outside the duodenal wall, usually forming a markedly long common channel. As the action of the sphincter of Oddi does not functionally affect the junction in PBM patients, continuous pancreatobiliary reflux occurs, resulting in a high incidence of biliary cancer. PBM can be divided into PBM with biliary dilatation (congenital choledochal cyst) and PBM without biliary dilatation (maximal diameter of the bile duct ≤ 10 mm). The treatment of choice for PBM is prophylactic surgery before malignant changes can take place. Endoscopic retrograde cholangiopancreatography (ERC P) is the most effective examination method for close obs ervation of the pattern of the junction site. When the communication between the pancreatic and bile ducts is maintained, despite contraction of the sphi ncter on ERCP, PBM is diagnosed. In these pat ients, levels of pancreatic enzymes in the bile are gene rally elevated, due to continuous pancreatobiliary reflux via a long common channel. Magnetic resonance cholangiopancreatography and 3D-computed tomography can diagnose PBM, based on findings of an anomalous union between the common bile duct and the pancreatic duct, in addition to a long common channel. Endoscopic ultrasonography and intraductal ultra sonography can demonstrate the junction outside the duodenal wall, and are useful for the diagnosis of asso ciated biliary cancer. Gallbladder wall thickness on ultra so nography can be a screening test for PBM.

关键词： pancreaticobiliary maljunction Pancreatobiliary reflux Congenital choledochal cyst Endoscopic retrograde cholangiopancreatography Endoscopic ultrasonography Magnetic resonance cholangiopancreatography

Endoscopic ultrasonography guided-fine needle aspirationfor the diagnosis of solid pancreaticobiliary lesions:Clinicalaspects to improve the diagnosis

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World Journal of Gastroenterology 2016年第2期22卷 628-640页

作者： Hiroyuki Matsubayashi Toru Matsui Yohei Yabuuchi Kenichiro Imai Masaki Tanaka Naomi Kakushima Keiko Sasaki Hiroyuki Ono Division of Endoscopy Shizuoka Cancer CenterSuntogunShizuoka 411-8777Japan Division of Pathology Shizuoka Cancer CenterSuntogunShizuoka 411-8777Japan

Endoscopic ultrasonography-guided fine-needle aspiration(EUS-FNA) has been applied to pancreaticobiliary lesions since the 1990 s and is in widespread use throughout the world today. We used this method to confirm the pathological evidence of the pancreaticobiliary lesions and to perform suitable therapies. Complications of EUS-FNA are quite rare, but some of them are severe. Operators should master conventional EUS observation and experience a minimum of 20-30 cases of supervised EUS-FNA on non-pancreatic and pancreatic lesions before attempting solo EUSFNA. Studies conducted on pancreaticobiliary EUSFNA have focused on selection of suitable instruments(e.g., needle selection) and sampling techniques(e.g., fanning method, suction level, with or without a stylet, optimum number of passes). Today, the diagnostic ability of EUS-FNA is still improving; the detection of pancreatic cancer(PC) currently has a sensitivity of 90%-95% and specificity of 95%-100%. In addition to PC, a variety of rare pancreatic tumors can be discriminated by conducting immunohistochemistry on the FNA materials. A flexible, large caliber needle has been used to obtain a large piece of tissue, which can provide sufficient histological information to be helpful in classifying benign pancreatic lesions. EUSFNA can supply high diagnostic yields even for biliary lesions or peri-pancreaticobiliary lymph nodes. This review focuses on the clinical aspects of EUS-FNA in the pancreaticobiliary field, with the aim of providing information that can enable more accurate and efficient diagnosis.

关键词： Endoscopic ultrasonography-guided fineneedleaspiration Diagnosis pancreaticobiliary Pancreatic Cancer

Application of endoscopic retrograde cholangiopancreatography in the diagnosis and treatment of pancreaticobiliary disease

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Journal of Medical Colleges of PLA(China) 1999年第1期14卷 57-61页

作者：孙振兴许国铭李兆申谢苏庆王娜田青吴仁培姚银珍方裕强

Objective: To validate endoscopic retrograde cholangiopancreatography (ERCP) in the diagnosis andtreatment of pancreaticobiliary disease and to improve the methodology of ERCP. Methods: From January 1977 toJune 1998, Seven thousand two hundred and thirty eight patients who were suspected to have pancreaticobiliaryduct diseases were examined with ERCP. Those who had therapeutic indications received ERCP treatment such asdrainage, dilatation and lithotomy. All cases were reviewed retrospectively, and analyzed for the clinical value andcomplications of ERCP. Results: In 7 238 patienls who underwent 7 579 ERCPs, the total success rate was94. 8%, with a 75. 7% showing rate of pancreatic duct. and 89. 1 % of the binary duct, revealing 3 492 cases ofbinary duct disease, 570 cases of pancreatic duct disease. 821 cases of diverticula and 171 cases of fistula. of which921 cases were treated endoscopically. The rate of complications arising from diagnostic ERCP was 1. 01%.without a single death, the rate of complications arising from therapeutic ERCP was 1. 3 %, two patients died(0. 22 % ). Conclusion: ERCP has important clinical value in the diagnosis and treatment of pancreaticobiliary ductdisease.

关键词： endoscopic retrograde cholangiopancreatography: pancreaticobiliary disease endoscopic diagnosis endoscopic therapy complications

Clinical implications of accessory pancreatic duct

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World Journal of Gastroenterology 2010年第36期16卷 4499-4503页

作者： Terumi Kamisawa Kensuke Takuma Taku Tabata Naoto Egawa Department of Internal Medicine Tokyo Metropolitan Komagome Hospital3-18-22 HonkomagomeBunkyo-kuTokyo 113-8677Japan

The accessory pancreatic duct (APD) is the main drainage duct of the dorsal pancreatic bud in the embryo,entering the duodenum at the minor duodenal papilla (MIP).With the growth,the duct of the dorsal bud undergoes varying degrees of atrophy at the duodenal *** of the APD in 291 control cases was 43% as determined by dye-injection endoscopic retrograde *** of the APD in 46 patients with acute pancreatitis was only 17%,which was significantly lower than in control cases (P pancreaticobiliary maljunction (PBM) were divided into a normal duct group (80 patients) and a dorsal pancreatic duct (DPD) dominant group (11 patients).While 48 patients (60%) with biliary carcinoma (gallbladder carcinoma,n=42;bile duct carcinoma,n=6) were identified in PBM with a normal pancreatic duct system,only two cases of gallbladder carcinoma (18%) occurred in DPD-dominant patients (P < 0.05).Concentration of amylase in the bile of DPD dominance was signifi cantly lower than that of normal pancreatic duct system (75 403.5 ± 82 015.4 IU/L vs 278 157.0 ± 207 395.0 IU/L,P < 0.05).In PBM with DPD dominance,most pancreatic juice in the upper DPD is drained into the duodenum via the MIP,and reflux of pancreatic juice to the biliary tract might be reduced,resulting in less frequency of associated biliary carcinoma.

关键词： Accessory pancreatic duct Minor duodenal papilla Pancreas divisum Main pancreatic duct Acute pancreatitis pancreaticobiliary maljunction

Choledochal cysts: Similarities and differences between Asian and Western countries

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World Journal of Gastroenterology 2019年第26期25卷 3334-3343页

作者： George N Baison Morgan M Bonds William S Helton Richard A Kozarek Digestive Disease Institute Virginia Mason Medical Center

Choledochal cysts(CCs)are rare bile duct dilatations,intra-and/or extrahepatic,and have higher prevalence in the Asian population compared to Western *** of the current literature on CC disease originates from Asia where these entities are most *** are thought to arise from an anomalous pancreaticobiliary junction,which are congenital anomalies between pancreatic and bile *** similarities in presentation between Eastern and Western patients exist such as female predominance,however,contemporary studies suggest that Asian patients may be more symptomatic on *** though CC disease presents with an increased malignant risk reported to be more than 10%after the second decade of life in Asian patients,this risk may be overstated in Western *** this difference in cancer risk,management guidelines for all patients with CC are based predominantly on observations reported from Asia where it is recommended that all CCs should be excised out of concern for the presence or development of biliary tract cancer.

关键词： Choledochal cyst Cholangiocarcinoma Asian populations Western populations Anomalous pancreaticobiliary junction