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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

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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

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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

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Metachronous cancer of gallbladder and pancreas with pancreatobiliary maljunction

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World Journal of Gastrointestinal Surgery 2010年第4期2卷 143-146页

作者： Ahlem Lahmar Sadri Ben Abid Mohamed Nafaa Arfa Rached Bayar Mohamed Tahar Khalfallah Sabah Mzabi-Regaya Department of Pathology Mongi Slim Hospital2046 Sidi-Daoued La MarsaTunisTunisia Department of Surgery and Liver Transplantation Mongi Slim Hospital2046 Sidi-Daoued La MarsaTunisTunisia

Pancreaticobiliary maljunction is a congenital anomaly in which the junction between the pancreatic duct and the common bile duct is located outside the sphincter of *** is well known that pancreaticobiliary maljunction is frequently associated with carcinoma of the biliary *** report a case of metachronous cancer of the gallbladder and pancreas associated with pancreaticobiliary maljunction and cystic dilatation of common bile duct in a 68-year-old Tunisian woman who underwent a cholecystectomy for acute *** pancreatic tumor was an adenosquamous *** maljunction allows for pancreatobiliary or biliopancreatic reflux which may induce biliary tract *** cases of multifocal cancer associated with this anomaly have been *** association with pancreatic carcinoma remains *** attention should be given to both the biliary tract system and pancreas during the long-term follow-up of patients with pancreaticobiliary maljunction,especially after they have undergone a choledochojejunostomy.

关键词： Pancreaticobiliary maljunction Gallbladder cancer Pancreatic cancer

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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

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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 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

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Pancreatic duct drainage using EUS-guided rendezvous technique for stenotic pancreaticojejunostomy

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World Journal of Gastroenterology 2013年第31期19卷 5182-5186页

作者： Tetsuya Takikawa Atsushi Kanno Atsushi Masamune Shin Hamada Eriko Nakano Shin Miura Hiroyuki Ariga Jun Unno Kiyoshi Kume Kazuhiro Kikuta Morihisa Hirota Hiroshi Yoshida Yu Katayose Michiaki Unno Tooru Shimosegawa Division of Gastroenterology Tohoku University Graduate School of Medicine Miyagi 980-8574 Japan Division of Hepato-biliary-Pancreatic Surgery Tohoku University Graduate School of Medicine Miyagi 980-8574 Japan

The patient was a 30-year-old female who had undergone excision of the extrahepatic bile duct and Rouxen-Y hepaticojejunostomy for congenital biliary dilatation at the age of ***,she suffered from recurrent acute pancreatitis due to pancreaticobiliary maljunction and received subtotal stomach-preserving *** developed a pancreatic fistula and an intra-abdominal abscess after the *** complications were improved by percutaneous abscess drainage and antibiotic *** ever,upper abdominal discomfort and the elevation of serum pancreatic enzymes persisted due to stenosis from the *** we could not accomplish dilation of the stenosis by endoscopic retrograde cholangiopancreatography,we tried an endoscopic ultrasonography(EUS) guided rendezvous technique for pancreatic duct *** transgastric puncture of the pancreatic duct using an EUS-fine needle aspiration needle,the guidewire was inserted into the pancreatic duct and finally reached to the jejunum through the stenotic *** changed the echoendoscope to an oblique-viewing endoscope,then grasped the guidewire and withdrew it through the *** stenosis of the pancreaticojejunostomy was dilated up to 4 mm,and a pancreatic stent was put in *** the pancreatic stent was removed after three months,the patient remained *** duct drainage using an EUS-guided rendezvous technique was useful for the treatment of a stenotic pancreaticojejunostomy after pancreaticoduodenectomy.

关键词： Balloon dilatation Endoscopic ultrasoundguided fine needle aspiration Pancreaticobiliary maljunction Pancreaticoduodenectomy Pancreatitis Postoperative complication

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Cholangiocarcinoma after flow diversion surgery for congenital biliary dilatation: A case report and review of literature

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World Journal of Hepatology 2019年第11期11卷 743-751页

作者： Ryo Ataka Takashi Ito Toshihiko Masui Satoru Seo Takamichi Ishii Satoshi Ogiso Shintaro Yagi Kojiro Taura Shinji Uemoto Department of Hepato-Biliary-Pancreatic Surgery and Transplantation Graduate School of Medicine Kyoto University

BACKGROUND Pancreaticobiliary maljunction(PBM)can be classified into two categories,PBM with congenital biliary dilatation(CBD)or PBM without biliary dilatation,and the management of PBM is often *** treatment for PBM with CBD is prophylactic flow diversion surgery,and some authors have reported that the incidence of cancer after extrahepatic bile duct excision is less than 1%.A very rare case of intrahepatic cholangiocarcinoma 6 years after flow diversion surgery for PBM with CBD is *** SUMMARY A 30-year-old man was diagnosed as having PBM with CBD,Todani classification type IVA,because of abnormal liver enzyme *** underwent flow diversion surgery and cholecystectomy,and the specimen showed adenocarcinoma foci,pT1,pStage *** and a half years passed without any recurrence of bile duct ***,6 years after his operation,computed tomography showed a gradually growing nodule in the bile *** positron emission tomography showed high uptake,and magnetic resonance imaging showed restricted diffusion *** double balloon enteroscopy,the nodule at the posterior bile duct-jejunum anastomosis was directly visualized,and its biopsy specimen showed *** patient underwent right lobectomy and biliary *** pathological diagnosis was intraductal papillary neoplasm with high-grade intraepithelial neoplasia,pTis,pN0,pStage *** patient’s postoperative course was uneventful,and he has had no recurrence up to the present *** This case suggests the necessity of careful observation after flow diversion surgery,especially when PBM with CBD is detected in adulthood.

关键词： Congenital biliary dilatation Pancreaticobiliary maljunction Cholangiocarcinoma Recurrence Metachronous neoplasm Case report

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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

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