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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摘要
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 condition.Here, 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 duct.CASE 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 pain.CONCLUSION 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.
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 condition.Here, 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 duct.CASE 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 pain.CONCLUSION 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.
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 condition.Here, 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 duct.CASE 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 pain.CONCLUSION 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.
引文
1 Kamisawa T,Ando H,Hamada Y,Fujii H,Koshinaga T,Urushihara N,Itoi T,Shimada H;Japanese Study Group on Pancreaticobiliary Maljunction.Diagnostic criteria for pancreaticobiliary maljunction2013.J Hepatobiliary Pancreat Sci 2014;21:159-161[PMID:24307541 DOI:10.1002/jhbp.57]
2 Babbitt DP,Starshak RJ,Clemett AR.Choledochal cyst:a concept of etiology.Am J Roentgenol Radium Ther Nucl Med 1973;119:57-62[PMID:4744730 DOI:10.2214/ajr.119.1.57]
3 Funabiki T,Matsubara T,Miyakawa S,Ishihara S.Pancreaticobiliary maljunction and carcinogenesis to biliary and pancreatic malignancy.Langenbecks Arch Surg 2009;394:159-169[PMID:18500533 DOI:10.1007/s00423-008-0336-0]
4 Komi N,Takehara H,Kunitomo K,Miyoshi Y,Yagi T.Does the type of anomalous arrangement of pancreaticobiliary ducts influence the surgery and prognosis of choledochal cyst?J Pediatr Surg 1992;27:728-731[PMID:1306647 DOI:10.1016/S0022-3468(05)80102-2]
5 Klein SD,Affronti JP.Pancreas divisum,an evidence-based review:part I,pathophysiology.Gastrointest Endosc 2004;60:419-425[PMID:15332034 DOI:10.1016/S0016-5107(04)01815-2]
6 Csendes A,Kruse A,Funch-Jensen P,Oster MJ,Ornsholt J,Amdrup E.Pressure measurements in the biliary and pancreatic duct systems in controls and in patients with gallstones,previous cholecystectomy,or common bile duct stones.Gastroenterology 1979;77:1203-1210[PMID:499707]
7 Terui K,Hishiki T,Saito T,Sato Y,Takenouchi A,Saito E,Ono S,Kamata T,Yoshida H.Pancreas divisum in pancreaticobiliary maljunction in children.Pediatr Surg Int 2010;26:419-422[PMID:20140733 DOI:10.1007/s00383-010-2559-8]
8 Matsumoto Y,Fujii H,Itakura J,Mogaki M,Matsuda M,Morozumi A,Fujino MA,Suda K.Pancreaticobiliary maljunction:etiologic concepts based on radiologic aspects.Gastrointest Endosc 2001;53:614-619[PMID:11323587 DOI:10.1067/mge.2001.113920]
9 Parlak E,K?ksal A?,Eminler AI.New type of anomalous pancreaticobiliary junction in a patient w?th choledochal cyst.Dig Endosc 2015;27:715[PMID:26081299 DOI:10.1111/den.12502]
10 Zhang Y,Sun W,Zhang F,Huang J,Fan Z.Pancreaticobiliary maljuction combining with pancreas divisum:Report of four cases.Exp Ther Med 2014;7:8-10[PMID:24348755 DOI:10.3892/etm.2013.1403]
11 McMahon CJ,Vollmer CM,Goldsmith J,Brown A,Pleskow D,Pedrosa I.An unusual variant of anomalous pancreaticobiliary junction in a patient with pancreas divisum diagnosed with secretin-magnetic resonance cholangiopancreatography.Pancreas 2010;39:101-104[PMID:20019565 DOI:10.1097/MPA.0b013e3181bd5eef]
12 De Angelis P,Foschia F,Romeo E,Caldaro T,Rea F,di Abriola GF,Caccamo R,Santi MR,Torroni F,Monti L,Dall'Oglio L.Role of endoscopic retrograde cholangiopancreatography in diagnosis and management of congenital choledochal cysts:28 pediatric cases.J Pediatr Surg 2012;47:885-888[PMID:22595566 DOI:10.1016/j.jpedsurg.2012.01.040]
13 Saito T,Terui K,Mitsunaga T,Nakata M,Kuriyama Y,Higashimoto Y,Kouchi K,Onuma N,Takahashi H,Yoshida H.Role of pediatric endoscopic retrograde cholangiopancreatography in an era stressing lessinvasive imaging modalities.J Pediatr Gastroenterol Nutr 2014;59:204-209[PMID:24762457 DOI:10.1097/MPG.0000000000000399]
14 Hiramatsu T,Itoh A,Kawashima H,Ohno E,Itoh Y,Sugimoto H,Sumi H,Funasaka K,Nakamura M,Miyahara R,Katano Y,Ishigami M,Ohmiya N,Kaneko K,Ando H,Goto H,Hirooka Y.Usefulness and safety of endoscopic retrograde cholangiopancreatography in children with pancreaticobiliary maljunction.J Pediatr Surg 2015;50:377-381[PMID:25746692 DOI:10.1016/j.jpedsurg.2014.08.024]
15 Carnes ML,Romagnuolo J,Cotton PB.Miss rate of pancreas divisum by magnetic resonance cholangiopancreatography in clinical practice.Pancreas 2008;37:151-153[PMID:18665075 DOI:10.1097/MPA.0b013e318164cbaf]
16 Kamisawa T,Tu Y,Egawa N,Tsuruta K,Okamoto A,Kamata N.MRCP of congenital pancreaticobiliary malformation.Abdom Imaging 2007;32:129-133[PMID:16680507 DOI:10.1007/s00261-006-9005-3]
17 Iwai N,Fumino S,Tsuda T,Ono S,Kimura O,Deguchi E.Surgical treatment for anomalous arrangement of the pancreaticobiliary duct with nondilatation of the common bile duct.J Pediatr Surg 2004;39:1794-1796[PMID:15616932 DOI:10.1016/j.jpedsurg.2004.08.010]
18 Ono S,Tokiwa K,Iwai N.Cellular activity in the gallbladder of children with anomalous arrangement of the pancreaticobiliary duct.J Pediatr Surg 1999;34:962-966[PMID:10392914 DOI:10.1016/S0022-3468(99)90769-8]
19 I??k A,Grassi A,Soran A.Positive Axilla in Breast Cancer;Clinical Practice in 2018.Eur J Breast Health2018;14:134-135[PMID:30123877 DOI:10.5152/ejbh.2018.4132]
20 Isik A,Firat D,Yilmaz I,Peker K,Idiz O,Yilmaz B,Demiryilmaz I,Celebi F.A survey of current approaches to thyroid nodules and thyroid operations.Int J Surg 2018;54:100-104[PMID:29709542DOI:10.1016/j.ijsu.2018.04.037]
21 Samavedy R,Sherman S,Lehman GA.Endoscopic therapy in anomalous pancreatobiliary duct junction.Gastrointest Endosc 1999;50:623-627[PMID:10536316 DOI:10.1016/S0016-5107(99)80009-1]
2 Babbitt DP,Starshak RJ,Clemett AR.Choledochal cyst:a concept of etiology.Am J Roentgenol Radium Ther Nucl Med 1973;119:57-62[PMID:4744730 DOI:10.2214/ajr.119.1.57]
3 Funabiki T,Matsubara T,Miyakawa S,Ishihara S.Pancreaticobiliary maljunction and carcinogenesis to biliary and pancreatic malignancy.Langenbecks Arch Surg 2009;394:159-169[PMID:18500533 DOI:10.1007/s00423-008-0336-0]
4 Komi N,Takehara H,Kunitomo K,Miyoshi Y,Yagi T.Does the type of anomalous arrangement of pancreaticobiliary ducts influence the surgery and prognosis of choledochal cyst?J Pediatr Surg 1992;27:728-731[PMID:1306647 DOI:10.1016/S0022-3468(05)80102-2]
5 Klein SD,Affronti JP.Pancreas divisum,an evidence-based review:part I,pathophysiology.Gastrointest Endosc 2004;60:419-425[PMID:15332034 DOI:10.1016/S0016-5107(04)01815-2]
6 Csendes A,Kruse A,Funch-Jensen P,Oster MJ,Ornsholt J,Amdrup E.Pressure measurements in the biliary and pancreatic duct systems in controls and in patients with gallstones,previous cholecystectomy,or common bile duct stones.Gastroenterology 1979;77:1203-1210[PMID:499707]
7 Terui K,Hishiki T,Saito T,Sato Y,Takenouchi A,Saito E,Ono S,Kamata T,Yoshida H.Pancreas divisum in pancreaticobiliary maljunction in children.Pediatr Surg Int 2010;26:419-422[PMID:20140733 DOI:10.1007/s00383-010-2559-8]
8 Matsumoto Y,Fujii H,Itakura J,Mogaki M,Matsuda M,Morozumi A,Fujino MA,Suda K.Pancreaticobiliary maljunction:etiologic concepts based on radiologic aspects.Gastrointest Endosc 2001;53:614-619[PMID:11323587 DOI:10.1067/mge.2001.113920]
9 Parlak E,K?ksal A?,Eminler AI.New type of anomalous pancreaticobiliary junction in a patient w?th choledochal cyst.Dig Endosc 2015;27:715[PMID:26081299 DOI:10.1111/den.12502]
10 Zhang Y,Sun W,Zhang F,Huang J,Fan Z.Pancreaticobiliary maljuction combining with pancreas divisum:Report of four cases.Exp Ther Med 2014;7:8-10[PMID:24348755 DOI:10.3892/etm.2013.1403]
11 McMahon CJ,Vollmer CM,Goldsmith J,Brown A,Pleskow D,Pedrosa I.An unusual variant of anomalous pancreaticobiliary junction in a patient with pancreas divisum diagnosed with secretin-magnetic resonance cholangiopancreatography.Pancreas 2010;39:101-104[PMID:20019565 DOI:10.1097/MPA.0b013e3181bd5eef]
12 De Angelis P,Foschia F,Romeo E,Caldaro T,Rea F,di Abriola GF,Caccamo R,Santi MR,Torroni F,Monti L,Dall'Oglio L.Role of endoscopic retrograde cholangiopancreatography in diagnosis and management of congenital choledochal cysts:28 pediatric cases.J Pediatr Surg 2012;47:885-888[PMID:22595566 DOI:10.1016/j.jpedsurg.2012.01.040]
13 Saito T,Terui K,Mitsunaga T,Nakata M,Kuriyama Y,Higashimoto Y,Kouchi K,Onuma N,Takahashi H,Yoshida H.Role of pediatric endoscopic retrograde cholangiopancreatography in an era stressing lessinvasive imaging modalities.J Pediatr Gastroenterol Nutr 2014;59:204-209[PMID:24762457 DOI:10.1097/MPG.0000000000000399]
14 Hiramatsu T,Itoh A,Kawashima H,Ohno E,Itoh Y,Sugimoto H,Sumi H,Funasaka K,Nakamura M,Miyahara R,Katano Y,Ishigami M,Ohmiya N,Kaneko K,Ando H,Goto H,Hirooka Y.Usefulness and safety of endoscopic retrograde cholangiopancreatography in children with pancreaticobiliary maljunction.J Pediatr Surg 2015;50:377-381[PMID:25746692 DOI:10.1016/j.jpedsurg.2014.08.024]
15 Carnes ML,Romagnuolo J,Cotton PB.Miss rate of pancreas divisum by magnetic resonance cholangiopancreatography in clinical practice.Pancreas 2008;37:151-153[PMID:18665075 DOI:10.1097/MPA.0b013e318164cbaf]
16 Kamisawa T,Tu Y,Egawa N,Tsuruta K,Okamoto A,Kamata N.MRCP of congenital pancreaticobiliary malformation.Abdom Imaging 2007;32:129-133[PMID:16680507 DOI:10.1007/s00261-006-9005-3]
17 Iwai N,Fumino S,Tsuda T,Ono S,Kimura O,Deguchi E.Surgical treatment for anomalous arrangement of the pancreaticobiliary duct with nondilatation of the common bile duct.J Pediatr Surg 2004;39:1794-1796[PMID:15616932 DOI:10.1016/j.jpedsurg.2004.08.010]
18 Ono S,Tokiwa K,Iwai N.Cellular activity in the gallbladder of children with anomalous arrangement of the pancreaticobiliary duct.J Pediatr Surg 1999;34:962-966[PMID:10392914 DOI:10.1016/S0022-3468(99)90769-8]
19 I??k A,Grassi A,Soran A.Positive Axilla in Breast Cancer;Clinical Practice in 2018.Eur J Breast Health2018;14:134-135[PMID:30123877 DOI:10.5152/ejbh.2018.4132]
20 Isik A,Firat D,Yilmaz I,Peker K,Idiz O,Yilmaz B,Demiryilmaz I,Celebi F.A survey of current approaches to thyroid nodules and thyroid operations.Int J Surg 2018;54:100-104[PMID:29709542DOI:10.1016/j.ijsu.2018.04.037]
21 Samavedy R,Sherman S,Lehman GA.Endoscopic therapy in anomalous pancreatobiliary duct junction.Gastrointest Endosc 1999;50:623-627[PMID:10536316 DOI:10.1016/S0016-5107(99)80009-1]