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1
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Mao HM, Chen KG, Zhu B, Guo WL, Shi SL. Deep learning radiomics nomogram for preoperatively identifying moderate-to-severe chronic cholangitis in children with pancreaticobiliary maljunction: a multicenter study. BMC Med Imaging 2025; 25:40. [PMID: 39910477 PMCID: PMC11800502 DOI: 10.1186/s12880-025-01579-3] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/07/2024] [Accepted: 02/02/2025] [Indexed: 02/07/2025] Open
Abstract
BACKGROUND Long-term severe cholangitis can lead to dense adhesions and increased fragility of the bile duct, complicating surgical procedures and elevating operative risk in children with pancreaticobiliary maljunction (PBM). Consequently, preoperative diagnosis of moderate-to-severe chronic cholangitis is essential for guiding treatment strategies and surgical planning. This study aimed to develop and validate a deep learning radiomics nomogram (DLRN) based on contrast-enhanced CT images and clinical characteristics to preoperatively identify moderate-to-severe chronic cholangitis in children with PBM. METHODS A total of 323 pediatric patients with PBM who underwent surgery were retrospectively enrolled from three centers, and divided into a training cohort (n = 153), an internal validation cohort (IVC, n = 67), and two external test cohorts (ETC1, n = 58; ETC2, n = 45). Chronic cholangitis severity was determined by postoperative pathology. Handcrafted radiomics features and deep learning (DL) radiomics features, extracted using transfer learning with the ResNet50 architecture, were obtained from portal venous-phase CT images. Multivariable logistic regression was used to establish the DLRN, integrating significant clinical factors with handcrafted and DL radiomics signatures. The diagnostic performances were evaluated in terms of discrimination, calibration, and clinical usefulness. RESULTS Biliary stones and peribiliary fluid collection were selected as important clinical factors. 5 handcrafted and 5 DL features were retained to build the two radiomics signatures, respectively. The integrated DLRN achieved satisfactory performance, achieving area under the curve (AUC) values of 0.913 (95% CI, 0.834-0.993), 0.916 (95% CI, 0.845-0.987), and 0.895 (95% CI, 0.801-0.989) in the IVC, and two ETCs, respectively. In comparison, the clinical model, handcrafted signature, and DL signature had AUC ranges of 0.654-0.705, 0.823-0.857, and 0.840-0.872 across the same cohorts. The DLRN outperformed single-modality clinical, handcrafted radiomics, and DL radiomics models, with all integrated discrimination improvement values > 0 and P < 0.05. The Hosmer-Lemeshow test and calibration curves showed good consistency of the DLRN (P > 0.05), and the decision curve analysis and clinical impact curve further confirmed its clinical utility. CONCLUSIONS The integrated DLRN can be a useful and non-invasive tool for preoperatively identifying moderate-to-severe chronic cholangitis in children with PBM, potentially enhancing clinical decision-making and personalized management strategies.
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Affiliation(s)
- Hui-Min Mao
- Department of Radiology, Children's Hospital of Soochow University, Suzhou, 215025, China
| | - Kai-Ge Chen
- Department of Ultrasound, Shandong Provincial Hospital Affiliated to Shandong First Medical University, Jinan, 250021, China
| | - Bin Zhu
- Department of Interventional Therapy, Xuzhou Children's Hospital, Xuzhou, 221000, China
| | - Wan-Liang Guo
- Department of Radiology, Children's Hospital of Soochow University, Suzhou, 215025, China.
| | - San-Li Shi
- Department of Radiology, The 8th Hospital of Xi'an, Xi'an, 710000, China.
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2
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Graterol FP, Marcano FS, Rivero-Moreno Y, Barrios YV. Komi type 2 pancreaticobiliary maljunction: Minimal access surgical treatment (with video). Ann Hepatobiliary Pancreat Surg 2024; 28:393-396. [PMID: 38867652 PMCID: PMC11341884 DOI: 10.14701/ahbps.24-063] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/12/2024] [Accepted: 04/18/2024] [Indexed: 06/14/2024] Open
Abstract
Pancreaticobiliary maljunction (PBM) is associated with the development of neoplasms of bile ducts. Cholecystectomy with diversion of the biliary-pancreatic flow is considered the treatment of choice. To describe the surgical treatment employed for a patient with Komi's type 2 PBM and its long-term results. Laparoscopic common bile duct exploration, intraoperative cholangioscopy, and Roux-en-Y hepatico-jejunostomy were performed. Postoperative evolution was satisfactory. The patient was discharge 72 hours after the surgery. There was no associated morbidity. At 62-month follow-up, clinical examination, laboratory tests, and imaging studies confirmed an adequate patency of bilio-enteric anastomosis. The surgical approach employed was effective and safe, with satisfactory long-term results.
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Korai T, Kimura Y, Watanabe K, Low SK, Imamura M, Nagayama M, Kukita K, Murakami T, Kato T, Kondo Y, Kyuno D, Sugawara T, Murota A, Kawakami Y, Masaki Y, Nakase H, Takemasa I. A case report of carcinoma of the papilla of Vater associated with a hyperplasia-dysplasia-carcinoma sequence by pancreaticobiliary maljunction. World J Surg Oncol 2024; 22:63. [PMID: 38389074 PMCID: PMC10882841 DOI: 10.1186/s12957-024-03347-z] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/30/2023] [Accepted: 02/17/2024] [Indexed: 02/24/2024] Open
Abstract
BACKGROUND Pancreaticobiliary maljunction (PBM) is a known risk factor for biliary tract cancer. However, its association with carcinoma of the papilla of Vater (PVca) remains unknown. We report a case with PVca that was thought to be caused by the hyperplasia-dysplasia-carcinoma sequence, which is considered a mechanism underlying PBM-induced biliary tract cancer. CASE PRESENTATION A 70-year-old woman presented with white stool and had a history of cholecystectomy for the diagnosis of a non-dilated biliary tract with PBM. Esophagogastroduodenoscopy revealed a tumor in the papilla of Vater, and PVca was histologically proven by biopsy. We finally diagnosed her with PVca concurrent with non-biliary dilated PBM (cT1aN0M0, cStage IA, according to the Union for International Cancer Control, 8th edition), and subsequently performed subtotal stomach-preserving pancreaticoduodenectomy. Pathological findings of the resected specimen revealed no adenomas and dysplastic and hyperplastic mucosae in the common channel slightly upstream of the main tumor, suggesting a PBM related carcinogenic pathway with hyperplasia-dysplasia-carcinoma sequence. Immunostaining revealed positivity for CEA. CK7 positivity, CK20 negativity, and MUC2 negativity indicated that this PVca was of the pancreatobiliary type. Genetic mutations were exclusively detected in tumors and not in normal tissues, and bile ducts from formalin-fixed paraffin-embedded samples included mutated-ERBB2 (Mutant allele frequency, 81.95%). Moreover, of the cell-free deoxyribonucleic acid (cfDNA) extracted from liquid biopsy mutated-ERBB2 was considered the circulating-tumor deoxyribonucleic acid (ctDNA) of this tumor. CONCLUSIONS Herein, we report the first case of PVca with PBM potentially caused by a "hyperplasia-dysplasia-carcinoma sequence" detected using immunostaining and next-generation sequencing. Careful follow-up is required if pancreaticobiliary reflux persists, considering the possible development of PVca.
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Affiliation(s)
- Takahiro Korai
- Department of Surgery, Surgical Oncology and Science, Sapporo Medical University School of Medicine, 291 Minami-1-jo Nishi 16-chome, Chuo-ku, Sapporo, Hokkaido, 060-8543, Japan
| | - Yasutoshi Kimura
- Department of Surgery, Surgical Oncology and Science, Sapporo Medical University School of Medicine, 291 Minami-1-jo Nishi 16-chome, Chuo-ku, Sapporo, Hokkaido, 060-8543, Japan.
| | - Kazunori Watanabe
- Hokkaido Gastroenterological Hospital, Sapporo, Hokkaido, Japan
- Department of Gastroenterological Surgery II, Faculty of Medicine, Hokkaido University, Sapporo, Hokkaido, Japan
| | - Siew-Kee Low
- Cancer Precision Medicine Center, Japanese Foundation for Cancer Research, Tokyo, Japan
| | - Masafumi Imamura
- Department of Surgery, Surgical Oncology and Science, Sapporo Medical University School of Medicine, 291 Minami-1-jo Nishi 16-chome, Chuo-ku, Sapporo, Hokkaido, 060-8543, Japan
| | - Minoru Nagayama
- Department of Surgery, Surgical Oncology and Science, Sapporo Medical University School of Medicine, 291 Minami-1-jo Nishi 16-chome, Chuo-ku, Sapporo, Hokkaido, 060-8543, Japan
| | - Kazuharu Kukita
- Department of Surgery, Surgical Oncology and Science, Sapporo Medical University School of Medicine, 291 Minami-1-jo Nishi 16-chome, Chuo-ku, Sapporo, Hokkaido, 060-8543, Japan
| | - Takeshi Murakami
- Department of Surgery, Surgical Oncology and Science, Sapporo Medical University School of Medicine, 291 Minami-1-jo Nishi 16-chome, Chuo-ku, Sapporo, Hokkaido, 060-8543, Japan
| | - Toru Kato
- Department of Surgery, Surgical Oncology and Science, Sapporo Medical University School of Medicine, 291 Minami-1-jo Nishi 16-chome, Chuo-ku, Sapporo, Hokkaido, 060-8543, Japan
| | - Yuta Kondo
- Department of Surgery, Surgical Oncology and Science, Sapporo Medical University School of Medicine, 291 Minami-1-jo Nishi 16-chome, Chuo-ku, Sapporo, Hokkaido, 060-8543, Japan
| | - Daisuke Kyuno
- Department of Surgery, Surgical Oncology and Science, Sapporo Medical University School of Medicine, 291 Minami-1-jo Nishi 16-chome, Chuo-ku, Sapporo, Hokkaido, 060-8543, Japan
| | - Taro Sugawara
- Department of Surgical Pathology, Sapporo Medical University School of Medicine, 291 Minami-1-jo Nishi 16-chome, Chuo-ku, Sapporo, 060-8543, Hokkaido, Japan
| | - Ayako Murota
- Department of Gastroenterology and Hepatology, Sapporo Medical University School of Medicine, Sapporo, Hokkaido, Japan
| | - Yujiro Kawakami
- Department of Gastroenterology and Hepatology, Sapporo Medical University School of Medicine, Sapporo, Hokkaido, Japan
| | - Yoshiharu Masaki
- Department of Gastroenterology and Hepatology, Sapporo Medical University School of Medicine, Sapporo, Hokkaido, Japan
| | - Hiroshi Nakase
- Department of Gastroenterology and Hepatology, Sapporo Medical University School of Medicine, Sapporo, Hokkaido, Japan
| | - Ichiro Takemasa
- Department of Surgery, Surgical Oncology and Science, Sapporo Medical University School of Medicine, 291 Minami-1-jo Nishi 16-chome, Chuo-ku, Sapporo, Hokkaido, 060-8543, Japan
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Xu Q, Liu M, Wu Q, Ling W, Guo S. Retrospective Analysis of the Accuracy of High-Frequency Ultrasound for Pancreaticobiliary Maljunction in Pediatrics at a Single Center. Front Pediatr 2022; 10:775378. [PMID: 35498787 PMCID: PMC9047754 DOI: 10.3389/fped.2022.775378] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/13/2021] [Accepted: 02/08/2022] [Indexed: 11/18/2022] Open
Abstract
OBJECTIVE To determine the clinical value of high-frequency ultrasonography (US) in the evaluation and diagnosis of pancreaticobiliary maljunction (PBM) among children. METHODS The clinical subjects consisted of 31 pediatric patients who were diagnosed with PBM from January 2015 to May 2021 in Fujian Provincial Maternity and Children's Hospital. The primary outcomes included diagnosis accuracy, imaging characteristics of each type of PBM based on JSPBM, time length of operation, and cost of service. Secondary outcomes were the serum amylase and bilirubin levels. RESULTS The diagnostic accuracy of US was 90.3% and comparable to the other imaging methods-MRCP (82.6%), IOC (79.2%), and ERCP (100%), respectively. The time length of operation and direct cost were significantly lower than other imaging pathways. Stenotic type (A) is associated with a high internal diameter of CBD, and dilated channel type (C) presents increased internal diameter and length of CC as well as internal diameter of PD. There were higher levels of the serum bilirubin seen in type A and of serum amylase in type C compared with others. CONCLUSION High-frequency US is a safe, cost-effective, and non-invasive imaging tool for the diagnosis and evaluation of PBM in pediatrics.
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Affiliation(s)
- Qiuchen Xu
- Department of Medical Ultrasonics, Fujian Maternity and Child Health Hospital, Affiliated Hospital of Fujian Medical University, Fuzhou, China
| | - Min Liu
- Department of Medical Ultrasonics, Fujian Maternity and Child Health Hospital, Affiliated Hospital of Fujian Medical University, Fuzhou, China
| | - Qiumei Wu
- Department of Medical Ultrasonics, Fujian Maternity and Child Health Hospital, Affiliated Hospital of Fujian Medical University, Fuzhou, China
| | - Wen Ling
- Department of Medical Ultrasonics, Fujian Maternity and Child Health Hospital, Affiliated Hospital of Fujian Medical University, Fuzhou, China
| | - Shan Guo
- Department of Medical Ultrasonics, Fujian Maternity and Child Health Hospital, Affiliated Hospital of Fujian Medical University, Fuzhou, China
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5
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Liu F, Lan M, Xu X, Tao B, Chang X, Ye Z, Zeng J. Application of Embedding Hepaticojejunostomy in Children with Pancreaticobiliary Maljunction Without Biliary Dilatation. J Laparoendosc Adv Surg Tech A 2021; 32:336-341. [PMID: 34748413 DOI: 10.1089/lap.2021.0634] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/12/2022] Open
Abstract
Background: To investigate the effect of embedded hepaticojejunostomy in children with pancreaticobiliary maljunction (PBM) without biliary dilatation. Materials and Methods: The clinical data of 10 patients with nondilated PBM from February 2017 to July 2020 were retrospectively analyzed. Perioperative liver function indexes were compared. Results: All patients were diagnosed by magnetic resonance cholangiopancreatography (MRCP) combined with intraoperative cholangiography. There were 5 cases of Komi type I and 5 cases of type II; the diameter of the common bile duct was 4-9 mm (median: 6 mm); and the length of the common channel was 5-15 mm (median: 9.25 mm). The procedure for one patient with common duct stones was converted to open surgery. Laparoscopic cholecystectomy, common bile duct resection, and embedded hepaticojejunostomy were successfully performed in all 10 cases. The average operation time was 225 ± 96.64 min, and the intraoperative blood loss was 2-5 mL. The mean time to oral intake was 3.5 ± 1.65 days (range: 2-5 days), and the mean hospitalization duration was 6.2 ± 2.44 days (range: 5-8 days). The differences in liver function indexes in the perioperative period were statistically significant (P < .05). The patients were followed-up for 13 to 54 months (median: 40 months). All patients grew well and there was no bile duct dilatation, calculus, or cirrhosis on B-ultrasound examinations. Conclusions: The clinical manifestations of nondilated PBM are often concealed, and preoperative MRCP was important for obtaining a diagnosis. Laparoscopic cholecystectomy, common bile duct resection, and embedded hepaticojejunostomy are feasible for treating nondilated PBM.
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Affiliation(s)
- Fei Liu
- Department of Pediatric Surgery, Guangzhou Institute of Pediatrics, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, China
| | - Menglong Lan
- Department of Pediatric Surgery, Guangzhou Institute of Pediatrics, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, China
| | - Xiaogang Xu
- Department of Pediatric Surgery, Guangzhou Institute of Pediatrics, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, China
| | - Boyuan Tao
- Department of Pediatric Surgery, Guangzhou Institute of Pediatrics, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, China
| | - Xiaopan Chang
- Department of Pediatric Surgery, Guangzhou Institute of Pediatrics, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, China
| | - Zhihua Ye
- Department of Pediatric Surgery, Guangzhou Institute of Pediatrics, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, China
| | - Jixiao Zeng
- Department of Pediatric Surgery, Guangzhou Institute of Pediatrics, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, China
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6
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Razumovsky AY, Shubin NV. [Surgical treatment of common bile duct malformations in children]. Khirurgiia (Mosk) 2021:92-97. [PMID: 33759476 DOI: 10.17116/hirurgia202104192] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/17/2022]
Abstract
The review is devoted to the current classifications of common bile duct malformations, authors' opinion regarding surgical approach and reconstruction of the bile ducts. The authors describe the modern modifications of bile duct reconstructions and estimate their effectiveness. PubMed database, Google Scholar and National Scientific Electronic Library eLIBRARY.ru databases were used. We analyzed the complications in children depending on various reconstructions, anatomical form, timing of reconstructive surgery and early diagnosis of this pathology.
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Affiliation(s)
- A Yu Razumovsky
- Pirogov Russian National Research Medical University, Moscow, Russia.,Filatov Children's City Clinical Hospital, Moscow, Russia
| | - N V Shubin
- Pirogov Russian National Research Medical University, Moscow, Russia.,Filatov Children's City Clinical Hospital, Moscow, Russia
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7
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Suzuki M, Minowa K, Isayama H, Shimizu T. Acute recurrent and chronic pancreatitis in children. Pediatr Int 2021; 63:137-149. [PMID: 32745358 DOI: 10.1111/ped.14415] [Citation(s) in RCA: 8] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/13/2020] [Revised: 06/27/2020] [Accepted: 07/28/2020] [Indexed: 12/14/2022]
Abstract
Acute recurrent pancreatitis (ARP) is defined as two distinct episodes of acute pancreatitis (AP), whereas chronic pancreatitis (CP) is caused by persistent inflammation of the pancreas. In children they are caused by genetic mutations, autoimmune pancreatitis, congenital pancreatic abnormalities, and other conditions. Acute recurrent pancreatitis is frequently a precursor to CP, and both are thought to be on the same disease continuum. In particular, genetic factors are associated with early progression of ARP to CP. The diagnosis of CP, as in AP, is based on clinical findings, biochemical tests, and imaging studies. Findings of exocrine pancreatic dysfunction are also important in the diagnosis of CP. A step-up strategy has become increasingly standard for the treatment of patients with CP. This strategy starts with endoscopic treatment, such as pancreatic sphincterotomy and stenting, and progresses to surgery should endoscopic therapy fail or prove technically impossible. Non-opioid (e.g. ibuprofen / naproxen) and opioid (e.g. oxycodone) forms of analgesia are widely used in pediatric patients with AP or CP, whereas pancreatic enzyme replacement therapy may be beneficial for patients with abdominal pain, steatorrhea, and malnutrition. Despite the disparity in the age of onset, pediatric CP patients display some similarities to adults in terms of disease progress. To reduce the risk of developing pancreatic exocrine inefficiency, diabetes and pancreatic cancer in the future, clinicians need to be aware of the current diagnostic approach and treatment methods for ARP and CP and refer them to a pediatric gastroenterologist in a timely manner.
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Affiliation(s)
- Mitsuyoshi Suzuki
- Department of Pediatrics, Juntendo University Faculty of Medicine, Tokyo, Japan
| | - Kei Minowa
- Department of Pediatrics, Juntendo University Faculty of Medicine, Tokyo, Japan
| | - Hiroyuki Isayama
- Department of Gastroenterology, Juntendo University Graduate School of Medicine, Tokyo, Japan
| | - Toshiaki Shimizu
- Department of Pediatrics, Juntendo University Faculty of Medicine, Tokyo, Japan
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8
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Sugawa C, Culver A, Sundaresan N, Lucas CE. Congenital pancreaticobiliary anomalies in an urban medical center in the United States. JGH OPEN 2020; 4:1176-1182. [PMID: 33319053 PMCID: PMC7731818 DOI: 10.1002/jgh3.12418] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 08/27/2020] [Accepted: 09/06/2020] [Indexed: 11/17/2022]
Abstract
Background and Aim Pancreaticobiliary anomalies are rare and often present with cryptic signs and symptoms, thus delaying appropriate treatment. Methods Endoscopic retrograde cholangiopancreatography (ERCP) was used to define pancreaticobiliary anomalies. A retrospective review was performed of 5522 ERCPs conducted at a tertiary care center from 1972 to 2015. Results There were 249 (4.5%) patients with pancreaticobiliary anomalies, including 179 patients with pancreas divisum (PD), 44 patients with choledochal cyst (CC) (Todani's classification Type I: extrahepatic cyst 31, Type III; choledochocele 9, Type V: Caroli's disease 4), 20 patients with anomalous pancreaticobiliary ductal union (APDU), and 6 patients with other abnormalities. Of 179 patients with pancreas divisum, 8 (4.5%) required minor sphincterotomies for multiple unexplained acute pancreatitis. Of the 31, 15 (48%) Type I CC patients underwent an operation. In patients with Type III CC (choledochocele), seven of the nine were treated by endoscopic sphincterotomy, and two patients were treated by surgery. Four patients with Type V CC (Caroli's disease) were managed nonoperatively. Of the 20 patients with APDU, 8 (40%) required operative intervention. Six patients were found to have other anomalies: two with pancreas bifidum, one with a duplication of the gallbladder, one with a cystic duct diverticulum, one with an annular pancreas, and one with an abnormal cystic duct origin. These patients were treated based on their etiology. Conclusion Pancreaticobiliary anomalies are rare and can be defined using ERCP. The appreciation of these abnormalities is important for the proper diagnosis and treatment of these rare biliary and pancreatic disorders.
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Affiliation(s)
- Choichi Sugawa
- Michael and Marian Ilitch Department of Surgery Wayne State University School of Medicine Detroit, Michigan USA
| | - Ashley Culver
- Michael and Marian Ilitch Department of Surgery Wayne State University School of Medicine Detroit, Michigan USA
| | - Naresh Sundaresan
- Michael and Marian Ilitch Department of Surgery Wayne State University School of Medicine Detroit, Michigan USA
| | - Charles E Lucas
- Michael and Marian Ilitch Department of Surgery Wayne State University School of Medicine Detroit, Michigan USA
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9
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Guo WL, Geng J, Zhao JG, Fang F, Huang SG, Wang J. Gene expression profiling reveals upregulated FUT1 and MYBPC1 in children with pancreaticobiliary maljunction. Braz J Med Biol Res 2019; 52:e8522. [PMID: 31365696 PMCID: PMC6668958 DOI: 10.1590/1414-431x20198522] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/26/2019] [Accepted: 05/29/2019] [Indexed: 02/07/2023] Open
Abstract
Pancreaticobiliary maljunction (PBM) is associated with high risk of epithelial atypical growth and malignant transformation of the bile duct or gallbladder. However, overall changes in genetic expression have not been examined in children with PBM. Genome-wide expression was analyzed using peripheral blood samples from 10 children with PBM and 15 pediatric controls. Differentially expressed genes (DEGs) were identified using microarray. Bioinformatics analysis was conducted using Gene Ontology and KEGG analyses. The top 5 in the up-regulated genes in PBM were verified with qRT-PCR. Receiver operator characteristic curve analysis was conducted to evaluate the predictive accuracy of selected genes for PBM. The microarray experiments identified a total of 876 DEGs in PBM, among which 530 were up-regulated and the remaining 346 were down-regulated. Verification of the top 5 up-regulated genes (TYMS, MYBPC1, FUT1, XAGE2, and GREB1L) by qRT-PCR confirmed the up-regulation of MYBPC1 and FUT1. Receiver operating characteristic curve analysis suggested that FUT1 and MYBPC1 up-regulation could be used to predict PBM, with the area under the curve of 0.873 (95%CI=0.735-1.000) and 0.960 (95%CI=0.891-1.000), respectively. FUT1 and MYBPC1 were up-regulated in children with PBM, and could be used as potential biomarkers for PBM.
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Affiliation(s)
- Wan-Liang Guo
- Department of Radiology, Children's Hospital of Soochow University, Suzhou, China
| | - Jia Geng
- Clinical Laboratory, the 3rd Hospital of Yulin, Yulin, China
| | - Jun-gang Zhao
- Department of Pediatric Surgery, Children's Hospital of Soochow University, Suzhou, China
| | - Fang Fang
- Department of Pediatric Surgery, Children's Hospital of Soochow University, Suzhou, China
| | - Shun-Gen Huang
- Department of Pediatric Surgery, Children's Hospital of Soochow University, Suzhou, China
| | - Jian Wang
- Department of Pediatric Surgery, Children's Hospital of Soochow University, Suzhou, China
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10
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Baison GN, Bonds MM, Helton WS, Kozarek RA. Choledochal cysts: Similarities and differences between Asian and Western countries. World J Gastroenterol 2019; 25:3334-3343. [PMID: 31341359 PMCID: PMC6639560 DOI: 10.3748/wjg.v25.i26.3334] [Citation(s) in RCA: 35] [Impact Index Per Article: 5.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/15/2019] [Revised: 05/05/2019] [Accepted: 06/01/2019] [Indexed: 02/06/2023] Open
Abstract
Choledochal cysts (CCs) are rare bile duct dilatations, intra-and/or extrahepatic, and have higher prevalence in the Asian population compared to Western populations. Most of the current literature on CC disease originates from Asia where these entities are most prevalent. They are thought to arise from an anomalous pancreaticobiliary junction, which are congenital anomalies between pancreatic and bile ducts. Some 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 presentation. Even 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 populations. Despite 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.
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Affiliation(s)
- George N Baison
- Digestive Disease Institute, Virginia Mason Medical Center, Seattle, WA 98101, United States
| | - Morgan M Bonds
- Digestive Disease Institute, Virginia Mason Medical Center, Seattle, WA 98101, United States
| | - William S Helton
- Digestive Disease Institute, Virginia Mason Medical Center, Seattle, WA 98101, United States
| | - Richard A Kozarek
- Digestive Disease Institute, Virginia Mason Medical Center, Seattle, WA 98101, United States
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11
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Laparoscopic approach for choledochojejunostomy. Hepatobiliary Pancreat Dis Int 2019; 18:285-288. [PMID: 31023579 DOI: 10.1016/j.hbpd.2019.04.004] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/05/2018] [Accepted: 04/08/2019] [Indexed: 02/05/2023]
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12
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Friedmacher F, Ford KE, Davenport M. Choledochal malformations: global research, scientific advances and key controversies. Pediatr Surg Int 2019; 35:273-282. [PMID: 30406431 DOI: 10.1007/s00383-018-4392-4] [Citation(s) in RCA: 19] [Impact Index Per Article: 3.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 10/23/2018] [Indexed: 02/07/2023]
Abstract
Choledochal malformations (CMs) represent a spectrum of relatively rare and complex congenital anomalies, characterized by abnormal dilatation of the biliary tract in the absence of any acute obstruction. Today, almost 20% of CMs can be detected in-utero using maternal ultrasonography. Formal scientometric analysis was used to identify where modern CM research is taking place and perhaps where our attention should be directed in the future. Thus, this article offers a comprehensive review of recent scientific advances relating to CMs including the current understanding of etiology and classification, whilst also discussing key controversies such as risk of malignant transformation and the role of newer modalities of surgical treatment. Although laparoscopic excision of CMs and biliary reconstruction is nowadays feasible and safe, care should be taken before dispensing with standard open techniques, which have minimal complication rates and proven long-term benefit.
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Affiliation(s)
- Florian Friedmacher
- Department of Pediatric Surgery, King's College Hospital, Denmark Hill, London, SE5 9RS, UK. .,Department of Pediatric Surgery, The Royal London Hospital, London, UK.
| | - Kathryn E Ford
- Department of Pediatric Surgery, King's College Hospital, Denmark Hill, London, SE5 9RS, UK.,Department of Pediatric Surgery, Oxford University Hospitals, Oxford, UK
| | - Mark Davenport
- Department of Pediatric Surgery, King's College Hospital, Denmark Hill, London, SE5 9RS, UK
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Ando H, Takada T. Cystic Disorders of the Bile Ducts. SHACKELFORD'S SURGERY OF THE ALIMENTARY TRACT, 2 VOLUME SET 2019:1367-1377. [DOI: 10.1016/b978-0-323-40232-3.00116-3] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/03/2025]
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14
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Kamisawa T, Kaneko K, Itoi T, Ando H. Pancreaticobiliary maljunction and congenital biliary dilatation. Lancet Gastroenterol Hepatol 2018; 2:610-618. [PMID: 28691687 DOI: 10.1016/s2468-1253(17)30002-x] [Citation(s) in RCA: 70] [Impact Index Per Article: 10.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/17/2016] [Revised: 12/27/2016] [Accepted: 01/03/2017] [Indexed: 02/07/2023]
Abstract
Pancreaticobiliary maljunction is a congenital malformation in which the pancreatic and bile ducts join outside the duodenal wall, usually forming a long common channel. Because the action of the sphincter of Oddi does not regulate the function of the pancreaticobiliary junction in patients with pancreaticobiliary maljunction, two-way regurgitation occurs. Reflux of pancreatic juice into the biliary tract is associated with a high incidence of biliary cancer. Biliary carcinogenesis in patients with pancreaticobiliary maljunction is thought to follow the hyperplasia, dysplasia, then carcinoma sequence due to chronic inflammation caused by pancreatobiliary reflux. Pancreaticobiliary maljunction is diagnosed when an abnormally long common channel is evident on imaging studies. Congenital biliary dilatation involves both local dilatation of the extrahepatic bile duct, including the common bile duct, and pancreaticobiliary maljunction. Extrahepatic bile duct resection is the standard surgery for congenital biliary dilatation. However, complete excision of the intrapancreatic bile duct and removal of stenoses of the hepatic ducts are necessary to prevent serious complications after surgery.
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Affiliation(s)
- Terumi Kamisawa
- Department of Internal Medicine, Tokyo Metropolitan Komagome Hospital, Tokyo, Japan.
| | - Kenitiro Kaneko
- Division of Pediatric Surgery, Department of Surgery, Aichi Medical University, Nagakute, Japan
| | - Takao Itoi
- Department of Gastroenterology and Hepatology, Tokyo Medical University, Tokyo, Japan
| | - Hisami Ando
- Department of Pediatric Surgery, Aichi Prefectural Colony, Kasugai, Japan
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Kobayashi T, Ohashi T, Sakata J, Miura K, Wakai T. Cholangiocarcinoma Developing from the Remnant Bile Ducts Following Cyst Excision for Congenital Biliary Dilatation. PANCREATICOBILIARY MALJUNCTION AND CONGENITAL BILIARY DILATATION 2018:253-261. [DOI: 10.1007/978-981-10-8654-0_30] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/03/2025]
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Yamanaka T, Araki K, Ishii N, Tsukagoshi M, Igarashi T, Watanabe A, Kubo N, Kuwano H, Shirabe K. Carcinoma of the Papilla of Vater after Diversion Operation for Pancreaticobiliary Maljunction. Case Rep Gastroenterol 2017. [PMID: 28626371 PMCID: PMC5471824 DOI: 10.1159/000462967] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022] Open
Abstract
Pancreaticobiliary maljunction (PBM) is a congenital malformation that is associated with biliary cancer development. When patients are diagnosed with PBM, a diversion operation is recommended. Although a risk remains for developing residual bile duct carcinoma following diversion, the development of a carcinoma of the ampulla of Vater after a diversion operation for PBM is rare. We present a treated case of carcinoma of the ampulla of Vater after a diversion operation for PBM. A 65-year-old woman presented with abdominal pain. She had undergone extrahepatic bile duct resection and cholecystectomy 2 years 9 months previously for the treatment of type Ic PBM according to the Todani classification. At the current admission for evaluation of the abdominal pain, computed tomography and magnetic resonance imaging showed only dilation of the main pancreatic duct. However, gastrointestinal endoscopy showed a tumor at the papilla of Vater, and biopsy revealed adenocarcinoma of the papilla of Vater. We performed pylorus-preserving pancreaticoduodenectomy, and the pathological diagnosis was moderately differentiated tubular adenocarcinoma of the papilla of Vater with no metastasis to the lymph nodes. The patient remained in good health for 3 years postoperatively. Carcinoma of the papilla of Vater after a diversion operation for PBM is rare. In this case, a diagnosis could not be made by computed tomography or magnetic resonance imaging; the definitive diagnosis was obtained with gastrointestinal endoscopy. Careful postoperative follow-up with gastrointestinal endoscopy in addition to imaging examination may be needed after a diversion operation for PBM.
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Affiliation(s)
- Takahiro Yamanaka
- Department of Hepatobiliary and Pancreatic Surgery, Gunma University Graduate School of Medicine, Maebashi, Japan
| | - Kenichiro Araki
- Department of Hepatobiliary and Pancreatic Surgery, Gunma University Graduate School of Medicine, Maebashi, Japan
| | - Norihiro Ishii
- Department of Hepatobiliary and Pancreatic Surgery, Gunma University Graduate School of Medicine, Maebashi, Japan
| | - Mariko Tsukagoshi
- Department of Hepatobiliary and Pancreatic Surgery, Gunma University Graduate School of Medicine, Maebashi, Japan
| | - Takamichi Igarashi
- Department of Hepatobiliary and Pancreatic Surgery, Gunma University Graduate School of Medicine, Maebashi, Japan
| | - Akira Watanabe
- Department of Hepatobiliary and Pancreatic Surgery, Gunma University Graduate School of Medicine, Maebashi, Japan
| | - Norio Kubo
- Department of Hepatobiliary and Pancreatic Surgery, Gunma University Graduate School of Medicine, Maebashi, Japan
| | - Hiroyuki Kuwano
- Department of General Surgical Science (Surgery I), Gunma University Graduate School of Medicine, Maebashi, Japan
| | - Ken Shirabe
- Department of Hepatobiliary and Pancreatic Surgery, Gunma University Graduate School of Medicine, Maebashi, Japan
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Sugita R, Furuta A, Yamazaki T, Ito K, Noda Y. Pancreaticobiliary Juice Reflux in Patients With a Morphologically Normal Ductal System: Assessment Using Unenhanced MRI With Spin Labeling. AJR Am J Roentgenol 2017; 208:322-327. [DOI: 10.2214/ajr.16.16601] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 08/30/2023]
Affiliation(s)
- Reiji Sugita
- Department of Radiology, Sendai City Medical Center, 5-22-1, Tsurugaya, Miyagino-ku, Sendai, Miyagi 983-0824, Japan
| | - Akemi Furuta
- Department of Radiology, Ishinomaki Red Cross Hospital, Ishinomaki, Miyagi, Japan
| | - Tetsuro Yamazaki
- Department of Radiology, Sendai Kosei Hospital, Sendai, Miyagi, Japan
| | - Kei Ito
- Department of Gastroenterology, Sendai City Medical Center, Sendai, Miyagi, Japan
| | - Yutaka Noda
- Department of Gastroenterology, Sendai City Medical Center, Sendai, Miyagi, Japan
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Kamisawa T, Kuruma S, Chiba K, Tabata T, Koizumi S, Kikuyama M. Biliary carcinogenesis in pancreaticobiliary maljunction. J Gastroenterol 2017; 52:158-163. [PMID: 27704265 DOI: 10.1007/s00535-016-1268-z] [Citation(s) in RCA: 45] [Impact Index Per Article: 5.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/29/2016] [Accepted: 09/15/2016] [Indexed: 02/07/2023]
Abstract
Pancreaticobiliary maljunction (PBM) is a congenital malformation in which the pancreatic and bile ducts join anatomically outside the duodenal wall. Because of the excessive length of the common channel in PBM, sphincter action does not directly affect the pancreaticobiliary junction, which allows pancreatic juice to reflux into the biliary tract. According to the results of a nationwide survey, bile duct and gallbladder cancers were found in 6.9 and 13.4 % of adult patients with congenital biliary dilatation, respectively, and in 3.1 and 37.4 % of those with PBM without biliary dilatation, respectively. Biliary tract cancers develop about 15-20 years earlier in patients with PBM than in individuals without PBM; they sometimes develop as double cancers. Carcinogenesis is strongly associated with stasis of bile intermingled with refluxed pancreatic juice. Epithelial cells in the biliary tract of PBM patients are under constant attack from activated pancreatic enzymes, increased secondary bile acids, or other mutagens. This can result in hyperplastic change with increased cell proliferation activity, and in turn, oncogene and/or tumor suppressor gene mutations in the epithelia, leading to the biliary tract carcinogenesis. The carcinogenesis of biliary tract cancer accompanying PBM is considered to involve a hyperplasia-dysplasia-carcinoma sequence induced by chronic inflammation caused by the reflux of pancreatic juice into the biliary tract, which differs from the adenoma-carcinoma sequence or the de novo carcinogenesis associated with biliary tract cancers in the population without PBM. Patients with a relatively long common channel have a similar, albeit slightly lower, risk for gallbladder cancer compared with PBM patients.
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Affiliation(s)
- Terumi Kamisawa
- Department of Internal Medicine, Tokyo Metropolitan Komagome Hospital, 3-18-22 Honkomagome, Bunkyo-ku, Tokyo, 113-8677, Japan.
| | - Sawako Kuruma
- Department of Internal Medicine, Tokyo Metropolitan Komagome Hospital, 3-18-22 Honkomagome, Bunkyo-ku, Tokyo, 113-8677, Japan
| | - Kazuro Chiba
- Department of Internal Medicine, Tokyo Metropolitan Komagome Hospital, 3-18-22 Honkomagome, Bunkyo-ku, Tokyo, 113-8677, Japan
| | - Taku Tabata
- Department of Internal Medicine, Tokyo Metropolitan Komagome Hospital, 3-18-22 Honkomagome, Bunkyo-ku, Tokyo, 113-8677, Japan
| | - Satomi Koizumi
- Department of Internal Medicine, Tokyo Metropolitan Komagome Hospital, 3-18-22 Honkomagome, Bunkyo-ku, Tokyo, 113-8677, Japan
| | - Masataka Kikuyama
- Department of Internal Medicine, Tokyo Metropolitan Komagome Hospital, 3-18-22 Honkomagome, Bunkyo-ku, Tokyo, 113-8677, Japan
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Hamada Y, Ando H, Kamisawa T, Itoi T, Urushihara N, Koshinaga T, Saito T, Fujii H, Morotomi Y. Diagnostic criteria for congenital biliary dilatation 2015. JOURNAL OF HEPATO-BILIARY-PANCREATIC SCIENCES 2016; 23:342-6. [PMID: 26996969 DOI: 10.1002/jhbp.346] [Citation(s) in RCA: 66] [Impact Index Per Article: 7.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Received: 01/07/2016] [Accepted: 03/17/2016] [Indexed: 12/13/2022]
Abstract
BACKGROUND The Diagnostic Criteria for Pancreaticobiliary Maljunction 2013 were published by the Japanese Study Group on Pancreaticobiliary Maljunction (JSGPM) in 2014. The committee of JSGPM for diagnostic criteria for pancreaticobiliary maljunction has established the standard diameter of the bile duct, and a definition of dilatation of the bile duct was proposed in 2014. METHODS The committee of JSGPM prepared the diagnostic criteria for congenital biliary dilatation in 2014, and a final revised version was approved in 2015. RESULTS Congenital biliary dilatation is defined as a congenital malformation involving both local dilatation of the extrahepatic bile duct, including the common bile duct, and pancreaticobiliary maljunction. However, cases associated with intrahepatic bile duct dilatation can also be included. Various kinds of pathological conditions can occur on hepatobiliary systems and pancreas by bile duct dilatation and pancreaticobiliary maljunction. For a diagnosis of congenital biliary dilatation, both abnormal dilatation of the bile duct and pancreaticobiliary maljunction must be evident by either imaging test or anatomical examination. Acquired or secondary dilatation of the bile duct by obstruction due to biliary stones or malignancy should be strictly excluded. CONCLUSION Diagnostic criteria for congenital biliary dilatation 2015 were established from Japan representing a world first.
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Affiliation(s)
- Yoshinori Hamada
- Division of Pediatric Surgery, Department of Surgery, Kansai Medical University, Hirakata City, Osaka, Japan.
| | | | - Terumi Kamisawa
- Department of Internal Medicine, Tokyo Metropolitan Komagome Hospital, Tokyo, Japan
| | - Takao Itoi
- Department of Gastroenterology and Hepatology, Tokyo Medical University, Tokyo, Japan
| | - Naoto Urushihara
- Department of Pediatric Surgery, Shizuoka Children's Hospital, Shizuoka, Japan
| | - Tsugumichi Koshinaga
- Division of Pediatric Surgery, Department of Surgery, Nihon University School of Medicine, Tokyo, Japan
| | - Takeshi Saito
- Department of Pediatric Surgery, Chiba University School of Medicine, Chiba, Japan
| | - Hideki Fujii
- First Department of Surgery, University of Yamanashi, Kofu, Japan
| | - Yoshiki Morotomi
- Division of Pediatric Surgery, Department of Surgery, Osaka City University School of Medicine, Osaka, Japan
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Pancreaticobiliary maljunction and choledochal cysts: from embryogenesis to therapeutics aspects. Surg Radiol Anat 2016; 38:1053-1060. [DOI: 10.1007/s00276-016-1669-y] [Citation(s) in RCA: 16] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/09/2015] [Accepted: 03/10/2016] [Indexed: 02/07/2023]
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Sugita R. Pancreaticobiliary reflux as a high-risk factor for biliary malignancy: Clinical features and diagnostic advancements. World J Hepatol 2015; 7:1735-1741. [PMID: 26167246 PMCID: PMC4491902 DOI: 10.4254/wjh.v7.i13.1735] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/27/2015] [Revised: 03/18/2015] [Accepted: 04/20/2015] [Indexed: 02/06/2023] Open
Abstract
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 Oddi. Although the sphincter of Oddi functionally prevents the reflux of pancreatic juice, pancreaticobiliary reflux (PBR) occurs when function of the sphincter of Oddi halt. The anatomically abnormal junction is termed pancreaticobiliary maljunction (PBM) and is characterized by pancreatic and bile ducts joining outside of the duodenal wall. PBM 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 mucosa. On 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 attention. Although it has long been possible to diagnose PBM by various imaging modalities, PBR without PBM has remained difficult to assess. Therefore, the pathological features of PBR without PBM have not been yet fully elucidated. Lately, a new method of diagnosing PBR without PBM has appeared, and the features of PBR without PBM should soon be better understood.
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Anomalous union of the pancreaticobiliary duct without choledochal cyst: is cholecystectomy alone sufficient? Langenbecks Arch Surg 2015; 399:1071-6. [PMID: 25015305 DOI: 10.1007/s00423-014-1223-5] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/09/2014] [Accepted: 06/30/2014] [Indexed: 02/07/2023]
Abstract
PURPOSE Anomalous union of the pancreaticobiliary duct (AUPBD) is frequently associated with choledochal cyst and biliary tract cancers. Management of AUPBD with choledochal cyst consists of extrahepatic bile duct excision and cholecystectomy. In cases of AUPBD without choledochal cyst, cholecystectomy alone is usually recommended.This study aimed to evaluate the occurrence of biliary tract cancer in AUPBD patients in order to assess the validity of the currently recommended operative management. METHODS Of a total of 10,255 endoscopic retrograde cholangiopancreatography cases performed at two Korea University hospitals from 2001 to 2010, 55 (0.54 %) cases of AUPBD were identified. Patients with AUPBD were divided according to its subtype (P-C union and C-P union) and the presence of choledochal cyst for analysis. The occurrence of benign and malignant disease was evaluated and compared between the groups. RESULTS Gallbladder stones were more frequently found in AUPBD patients without choledochal cyst (p=0.032). Biliary tract cancer occurred more frequently in P-C union (p=0.050),especially the common bile duct cancer (p=0.023). When analyzed according to the presence of choledochal cyst, biliary tract cancer occurred more frequently in AUPBD patients without choledochal cyst (p=0.005), with bile duct cancer being significantly more common (p=0.015). However, there was no difference in the presence of gallbladder cancer between the two groups (p=0.318). CONCLUSIONS Since cancers of the biliary tract occur more frequently in the AUPBD group without choledochal cyst,cholecystectomy alone may not be protective of the future occurrence of bile duct cancers, and thus, vigilant surveillance is necessary in this population group.
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Kamisawa T, Kuruma S, Tabata T, Chiba K, Iwasaki S, Koizumi S, Kurata M, Honda G, Itoi T. Pancreaticobiliary maljunction and biliary cancer. J Gastroenterol 2015; 50:273-9. [PMID: 25404143 DOI: 10.1007/s00535-014-1015-2] [Citation(s) in RCA: 41] [Impact Index Per Article: 4.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/28/2014] [Accepted: 10/30/2014] [Indexed: 02/07/2023]
Abstract
Pancreaticobiliary maljunction (PBM) is a congenital malformation in which the pancreatic and bile ducts join anatomically outside the duodenal wall. Japanese clinical practice guidelines on how to deal with PBM were made in 2012, representing a world first. According to the 2013 revision to the diagnostic criteria for PBM, in addition to direct cholangiography, diagnosis can be made by magnetic resonance cholangiopancreatography (MRCP), 3-dimensional drip infusion cholangiography computed tomography, endoscopic ultrasonography (US), or multiplanar reconstruction images by multidetector row computed tomography. In PBM, the common channel is so long that sphincter action does not affect the pancreaticobiliary junction, and pancreatic juice frequently refluxes into the biliary tract. Persistence of refluxed pancreatic juice injures epithelium of the biliary tract and promotes cancer development, resulting in higher rates of carcinogenesis in the biliary tract. In a nationwide survey, biliary cancer was detected in 21.6% of adult patients with congenital biliary dilatation (bile duct cancer, 32.1% vs. gallbladder cancer, 62.3%) and in 42.4% of PBM patients without biliary dilatation (bile duct cancer, 7.3% vs. gallbladder cancer, 88.1%). Pathophysiological conditions due to pancreatobiliary reflux occur in patients with high confluence of pancreaticobiliary ducts, a common channel ≥6 mm long, and occlusion of communication during contraction of the sphincter. Once the diagnosis of PBM is established, immediate prophylactic surgery is recommended. However, the surgical strategy for PBM without biliary dilatation remains controversial. To detect PBM without biliary dilatation early, MRCP is recommended for patients showing gallbladder wall thickening on screening US under suspicion of PBM.
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Affiliation(s)
- Terumi Kamisawa
- Department of Internal Medicine, Tokyo Metropolitan Komagome Hospital, 3-18-22 Honkomagome, Bunkyo-ku, Tokyo, 113-8677, Japan,
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Chen F, Tang L, Zhang ZQ, Jin BW, Dong WF, Wang J, Huang SG. Liver, biliary and pancreatic injuries in pancreaticobiliary maljunction model in cats. Hepatobiliary Pancreat Dis Int 2015; 14:90-5. [PMID: 25655296 DOI: 10.1016/s1499-3872(14)60306-4] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/05/2023]
Abstract
BACKGROUND Pancreaticobiliary maljunction is a high risk factor of pancreatitis and biliary tract cancer. How this maljunction affects the liver remains obscure. This study aimed to examine the effects of pancreaticobiliary maljunction on the liver, pancreas and gallbladder in a cat model. METHODS A model of choledocho-pancreatic side-to-side ductal anastomosis was created in ten cats. Before the procedure, a small piece of tissue from the liver, pancreas and gallbladder was collected as a control. The common channel formation was checked by cholecystography. The livers, pancreases and gallbladders of these cats were harvested for histological examination. The expression of proliferating cell nuclear antigen in the gallbladder was examined with immunohistochemistry. RESULTS Seven of the 10 cats survived for 6 months after surgery. The color of the liver was darker in the PBM model than the control specimen, with nodules on the surface. Histological examination showed ballooning changes and inflammatory infiltrations and the histopathological score increased significantly (P<0.05). Also, mitochondria swelling and lipid droplet in cytoplasm were observed under an electron microscope. The pancreas also appeared darker in the PBM model than the control specimen and dilated pancreatic ducts were found in three cats. Histopathological examination revealed vascular proliferation and inflammatory infiltration with numerous neutrophils. Gallbladder epithelial cells were featured by expanded endoplasmic reticulum, increased intercellular space and cellular nucleus deformation. The positive cells of proliferating cell nuclear antigen were increased significantly (P<0.05). CONCLUSION The present study demonstrated that pancreaticobiliary maljunction can lead to the injuries of the liver, pancreas and gallbladder.
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Affiliation(s)
- Feng Chen
- Division of General Surgery, Soochow University Affiliated Children's Hospital, Suzhou 215003, China.
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Tang L, Wang XD, Xu JY, Wang J, Huang SG, Chen F. Anatomical and Morphological Features of the Fetal Human Pancreaticobiliary Ductal Union. Fetal Pediatr Pathol 2015; 34:355-60. [PMID: 26417940 DOI: 10.3109/15513815.2015.1087608] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/13/2022]
Abstract
To investigate pancreaticobiliary ductal anatomy during developmental stages, gallbladders, common bile ducts, pancreatic ducts and their interface with the duodenum were studied in 36 human fetuses between 4-6 weeks postconceptual age were studied. For histological examination, sections were cut continuously from the paraffin-embedded tissue block and stained with hematoxylin and eosin. The expression of proliferating cell nuclear antigen in the gallbladder was examined with immunohistochemistry. Among 36 cases, three shapes of the greater duodenal papilla were found: hemispheroid (58.1%), circular cylinder (25%), and flat shape (16.9%). For the location of the greater duodenal papillas, more than half (69.4%) of the cases were in the middle descendant duodenum. Seven cases (19.4%) were in the lower descendant duodenum. Three cases (8.3%) were in the upper descendant duodenum, and one (2.9%) was in the distal descending part of duodenum. There were four types of the pancreaticobiliary ductal union: "Y" in 24 cases(66.7%), "U" in 4 cases (11.1%),"V" in 7 cases (19.4%), and pancreaticobiliary maljunction in 1 case (2.8%). For patients with congenital bile duct dilation and Biliary cancer, the positive cells of proliferating cell nuclear antigen were increased significantly (P < 0.05). Different types in pancreaticobiliary ductal union investigated in this study may provide clues for pathogenesis and clinical treatment of pancreaticobiliary maljunction.
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Affiliation(s)
- Lin Tang
- a Nanjing University of Chinese Medicine , Nanjing , China
| | - Xing-Dong Wang
- b Soochow University Affiliated Children's Hospital , Suzhou , China
| | - Jian-Ya Xu
- b Soochow University Affiliated Children's Hospital , Suzhou , China
| | - Jian Wang
- b Soochow University Affiliated Children's Hospital , Suzhou , China
| | - Shun-Gen Huang
- b Soochow University Affiliated Children's Hospital , Suzhou , China
| | - Feng Chen
- c Shanghai Jiao Tong University Affiliated Sixth People's Hospital , Shanghai , China
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