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Benesch MG, Ramos-Santillan VO, Rog CJ, Nelson ED, Takabe K. Epidemiology of Adenosquamous Carcinomas. World J Oncol 2024; 15:432-453. [PMID: 38751700 PMCID: PMC11092416 DOI: 10.14740/wjon1845] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/07/2024] [Accepted: 04/15/2024] [Indexed: 05/18/2024] Open
Abstract
Background Adenosquamous carcinomas (ASCs) are a very rare histology containing cancer cells with both glandular-like (adeno) and squamous cell histologies, comprising typically a fraction of a percent of all solid tumors. The bulk of the literature on ASCs is comprised of case reports and small series, with the general finding that ASCs tend to have worse outcomes than either of their parent histologies. However, there is a lack of pan site-comparative studies in the literature that compare ASC clinicodemographic and survival outcomes with those of conventional adenocarcinomas (ACs) and squamous cell carcinomas (SCCs). Methods In this study, we summarize these outcomes in eight primary sites, comprising 92.7% of all ASC cases diagnosed from 1975 to 2020 in the Surveillance, Epidemiology, and End Results (SEER) database. Results Lung ASCs comprise 51.5% of all ASC cases, accounting for 1.1% of all lung cancer cases, followed by uterine/cervical cancers at 29.7% of all ASC cases, translating into 1.8% of all cancers in this site. In descending order, the remaining 20% of ASCs arise in pancreatic, oral cavity, biliary, esophageal, colorectal, and gastric sites, comprising between 0.1% and 0.7% of all cancers in these sites. Apart from pancreatic and oral cavity cancers, ASC tumors tended to favor higher rates of regional or distant disease at presentation with poor tumor differentiation compared to either AC or SCC histologies. After multivariable analysis, adjusting for age, sex, detection stage, grade differentiation, surgery, chemotherapy, and radiotherapy, except for oral cavity cancers, ASCs tended to have worse overall survivals compared to ACs (hazard ratios: 1.1 - 1.6) and SCC (1.0 - 1.3), with colorectal ASCs having the worse overall survival compared to colorectal ACs, with a hazard ratio of 1.4 (95% confidence interval: 1.3 - 1.6). Conclusions Overall, these results suggest that ASC outcomes are site specific, and in general, tend to have worse outcomes than nonvariant ACs and SCCs even after correction for common clinical and epidemiological factors. These cancers have a poorly understood but unique tumor biology that warrants further characterization.
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Affiliation(s)
- Matthew G.K. Benesch
- Department of Surgical Oncology, Roswell Park Comprehensive Cancer Center, Buffalo, NY 14263, USA
- These authors equally contributed to this work
| | - Vicente O. Ramos-Santillan
- Department of Surgical Oncology, Roswell Park Comprehensive Cancer Center, Buffalo, NY 14263, USA
- These authors equally contributed to this work
| | - Colin J. Rog
- Department of Surgical Oncology, Roswell Park Comprehensive Cancer Center, Buffalo, NY 14263, USA
| | - Erek D. Nelson
- Department of Surgical Oncology, Roswell Park Comprehensive Cancer Center, Buffalo, NY 14263, USA
| | - Kazuaki Takabe
- Department of Surgical Oncology, Roswell Park Comprehensive Cancer Center, Buffalo, NY 14263, USA
- Department of Breast Surgery and Oncology, Tokyo Medical University, Tokyo 160-8402, Japan
- Department of Gastroenterological Surgery, Yokohama City University Graduate School of Medicine, Yokohama 236-0004, Japan
- Division of Digestive and General Surgery, Niigata University Graduate School of Medical and Dental Sciences, Niigata 951-8520, Japan
- Department of Breast Surgery, Fukushima Medical University School of Medicine, Fukushima 960-1295, Japan
- Department of Surgery, University at Buffalo Jacobs School of Medicine and Biomedical Sciences, State University of New York, Buffalo, NY 14263, USA
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Diagnostic usefulness of SpyGlass in intracholecystic papillary neoplasm with pancreaticobiliary maljunction: a case report and comparison with conventional gallbladder cancer with pancreaticobiliary maljunction. Clin J Gastroenterol 2023; 16:476-481. [PMID: 36867354 DOI: 10.1007/s12328-023-01778-x] [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: 01/17/2023] [Accepted: 02/20/2023] [Indexed: 03/04/2023]
Abstract
BACKGROUND Intracholecystic papillary neoplasm (ICPN) is one of the precursors of gallbladder cancer defined in the 2010 World Health Organization classification of tumors. We herein report ICPN with pancreaticobiliary maljunction (PBM), which is a high-risk factor for biliary cancer. CASE PRESENTATION A 57-year-old female presented with abdominal pain. Computed tomography showed a swollen appendix and gallbladder nodules with bile duct dilatation. Endoscopic ultrasonography revealed a gallbladder tumor spreading into the cystic duct confluence accompanying PBM. Based on papillary tumors around the cystic duct detected using the SpyGlass DS II Direct Visualization System (SpyGlass DS), ICPN was suspected. We performed extended cholecystectomy, extrahepatic bile duct resection, and appendectomy with a diagnosis of ICPN and PBM. The pathological diagnosis was ICPN (90 × 50 mm) with high-grade dysplasia spreading into the common bile duct. The absence of residual cancer in the resected specimen was pathologically confirmed. P53 staining was totally negative in both the tumor and normal epithelium. The overexpression of CTNNB1 was not observed. CONCLUSIONS We encountered a patient with a very rare gallbladder tumor, ICPN with PBM. SpyGlass DS contributed to a precise assessment of the extent of the tumor as well as a qualitative diagnosis.
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Tidjane A, Boudjenan‐Serradj N, Khalifa S, Ikhlef N, Bengueddach A, Larbi H, Meharzi SEI, Tabeti B. Adenosquamous carcinoma of gallbladder associated with biliopancreatic maljunction and Todani 1c choledochal cyst: A Case Report. ADVANCES IN DIGESTIVE MEDICINE 2022. [DOI: 10.1002/aid2.13327] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/08/2022]
Affiliation(s)
- Anisse Tidjane
- Department of Hepatobiliary Surgery and Liver Transplantation, EHU‐1st November 1954 Oran Algeria
- Department of Medicine University of Oran 1 Oran Algeria
| | - Nabil Boudjenan‐Serradj
- Department of Hepatobiliary Surgery and Liver Transplantation, EHU‐1st November 1954 Oran Algeria
- Department of Medicine University of Oran 1 Oran Algeria
| | - Samia Khalifa
- Department of Histopathology, EHU‐1st November 1954 Oran Algeria
| | - Nacim Ikhlef
- Department of Hepatobiliary Surgery and Liver Transplantation, EHU‐1st November 1954 Oran Algeria
- Department of Medicine University of Oran 1 Oran Algeria
| | | | - Hakim Larbi
- Department of Hepatobiliary Surgery and Liver Transplantation, EHU‐1st November 1954 Oran Algeria
- Department of Medicine University of Oran 1 Oran Algeria
| | - Sif el Islam Meharzi
- Department of Hepatobiliary Surgery and Liver Transplantation, EHU‐1st November 1954 Oran Algeria
- Department of Medicine University of Oran 1 Oran Algeria
| | - Benali Tabeti
- Department of Hepatobiliary Surgery and Liver Transplantation, EHU‐1st November 1954 Oran Algeria
- Department of Medicine University of Oran 1 Oran Algeria
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Yoshikawa M, Ikemoto T, Morine Y, Imura S, Saito Y, Yamada S, Teraoku H, Takata A, Yoshimoto T, Shimada M. Aggressive resection of metachronous triple biliary cancer. THE JOURNAL OF MEDICAL INVESTIGATION 2018; 64:299-304. [PMID: 28955001 DOI: 10.2152/jmi.64.299] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/14/2022]
Abstract
Radical resection of recurrent cholangiocellular carcinoma (CCC) is rare. To date, only two patients have been reported to undergo major hepatectomy twice after pancreatoduodenectomy (PD) for metachronous CCC. This report describes a patient who underwent three metachronous radical resections of CCC with curative intent, consisting of PD followed by two hepatectomies, for cancer of the middle bile duct. A 60 year old male, who had undergone a distal gastrectomy for duodenal ulcer perforation in 23 years ago, underwent PD (Honjo method) for middle bile duct cancer (papillary adenocarcinoma) in 20 years ago at another hospital. In 12 years ago, he was referred to our hospital for further examinations of hepatic tumors and was diagnosed with CCC (S6, solitary, 4 cm). He underwent S6 subsegmentectomy of the liver. Pathological examination revealed a poorly differentiated CCC, considered a second primary cancer. In 6 years ago, subsequent follow up as an outpatient revealed repeated cholangitis, with CT showing a hilar mass caused by obstructive jaundice. He was diagnosed with a hilar cholangioma, and underwent hepatectomy and hepaticojejunostomy, which were safe despite surgical difficulties caused by severe adhesions and re-anastomosis. Pathological examination showed a well-differentiated CCC, considered the third primary cancer. The lung metastasis was revealed in 28 months after the last operation, however he has survived after partial lung resection without further metastasis. J. Med. Invest. 64: 299-304, August, 2017.
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Affiliation(s)
- Masato Yoshikawa
- Department of Surgery, Tokushima University Graduate Shool of Biomedical Sciences
| | - Tetsuya Ikemoto
- Department of Surgery, Tokushima University Graduate Shool of Biomedical Sciences
| | - Yuji Morine
- Department of Surgery, Tokushima University Graduate Shool of Biomedical Sciences
| | - Satoru Imura
- Department of Surgery, Tokushima University Graduate Shool of Biomedical Sciences
| | - Yu Saito
- Department of Surgery, Tokushima University Graduate Shool of Biomedical Sciences
| | - Shinichiro Yamada
- Department of Surgery, Tokushima University Graduate Shool of Biomedical Sciences
| | - Hiroki Teraoku
- Department of Surgery, Tokushima University Graduate Shool of Biomedical Sciences
| | - Atsushi Takata
- Department of Surgery, Tokushima University Graduate Shool of Biomedical Sciences
| | - Toshiaki Yoshimoto
- Department of Surgery, Tokushima University Graduate Shool of Biomedical Sciences
| | - Mitsuo Shimada
- Department of Surgery, Tokushima University Graduate Shool of Biomedical Sciences
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Qin BD, Jiao XD, Yuan LY, Liu K, Zang YS. Adenosquamous carcinoma of the bile duct: a population-based study. Cancer Manag Res 2018; 10:439-446. [PMID: 29563834 PMCID: PMC5846297 DOI: 10.2147/cmar.s144850] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/25/2022] Open
Abstract
Introduction Adenosquamous carcinoma (ASC) of the bile duct is a rare diagnosis with poorly understood clinicopathological characteristics and disease progression, so identification of the features associated with ASC patient survival is warranted. Materials and methods A population cohort study was performed using prospectively extracted data from the Surveillance, Epidemiology and End Results (SEER) database for patients with histological diagnoses of ASC of the bile duct from 1973 to 2013. Results A total of 106 patients with ASC of the bile duct were included (mean age 68.1±13.5 years). Lesions from 58 patients were in the extrahepatic bile duct and 34 were located at the ampulla of Vater. Fifty-seven patients were categorized with a regional stage, 15 had localized disease, and 30 had distant disease. Most (60.4%) patients received cancer-directed surgery, and radiation was performed in 14.1% of cases. The 1-year, 2-year, and 5-year overall survival (OS) for patients with ASC of the bile duct was 30.1%, 11.3%, and 3.7%, respectively. Cancer-directed surgery offered 10 additional months of OS for patients with ASC of the bile duct and median OS was 14.0, 6.0, and 6.0 months for ampulla of Vater, extrahepatic bile duct, and intrahepatic bile duct cases, respectively. A multivariate Cox analysis showed that lesions in the ampulla of Vater (HR=0.51, 95% CI 0.26–0.99) and having surgery (HR=0.34, 95% CI 0.14–0.81) were independent protective prognostic factors for these patients. Conclusion Cancer-directed surgery and a primary lesion site of the ampulla of Vater may suggest favorable prognosis for patients with ASC of the bile duct.
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Affiliation(s)
- Bao-Dong Qin
- Department of Medical Oncology, Changzheng Hospital, Second Military Medical University, Shanghai, China
| | - Xiao-Dong Jiao
- Department of Medical Oncology, Changzheng Hospital, Second Military Medical University, Shanghai, China
| | - Ling-Yan Yuan
- Department of Medical Oncology, Changzheng Hospital, Second Military Medical University, Shanghai, China
| | - Ke Liu
- Department of Medical Oncology, Changzheng Hospital, Second Military Medical University, Shanghai, China
| | - Yuan-Sheng Zang
- Department of Medical Oncology, Changzheng Hospital, Second Military Medical University, Shanghai, China
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Koizumi K, Sasajima J, Kawamoto T, Sugiyama Y, Muto M, Muto M, Ishikawa C, Inoue M, Kohgo Y. Multiple Cancers of the Biliary Tract and Pancreatic Duct after Cholecystectomy for Gallbladder Cancer in a Patient with Pancreaticobiliary Maljunction. Intern Med 2016; 55:141-6. [PMID: 26781013 DOI: 10.2169/internalmedicine.55.4706] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/06/2022] Open
Abstract
We herein report the rare case of a 76-year-old woman who underwent cholecystectomy with bile duct resection for advanced gallbladder cancer associated with pancreaticobiliary maljunction (PBM) and subsequently developed multiple cancers of the pancreaticobiliary system (the distal bile duct, intrahepatic duct and pancreatic duct) after the operation. We performed conventional endoscopic retrograde cholangiopancreatography (ERCP) using a side-viewing scope to evaluate the masses in the distal bile duct and the pancreatic duct. We also performed ERCP using double-balloon enteroscopy (DBE) to observe the mass in the intrahepatic duct. It was possible to directly observe the lesion using DBE and to perform a biopsy under visual control. All lesions were correctly diagnosed by the combination of ERCP using different endoscopes. The present case suggests that it is necessary to pay close attention (with regard to carcinogenesis) to the whole pancreaticobiliary system in patients with PBM. In addition, the combination of ERCP using DBE and a side-viewing scope may be useful for making a precise diagnosis in patients with altered biliary anatomy who have multiple cancers of the pancreaticobiliary system.
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Affiliation(s)
- Kazuya Koizumi
- Gastroenterology Medicine Center, Shonan Kamakura General Hospital, Japan
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8
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Kim DH, Choi DW, Choi SH, Heo JS. Pancreaticoduodenectomy for secondary periampullary cancer following extrahepatic bile duct cancer resection. Ann Surg Treat Res 2014; 87:94-9. [PMID: 25114889 PMCID: PMC4127899 DOI: 10.4174/astr.2014.87.2.94] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/07/2014] [Revised: 04/14/2014] [Accepted: 04/15/2014] [Indexed: 12/17/2022] Open
Abstract
PURPOSE This study addressed the feasibility and effect of surgical treatment of metachronous periampullary carcinoma after resection of the primary extrahepatic bile duct cancer. The performance of this secondary curative surgery is not well-documented. METHODS We reviewed, retrospectively, the medical records of 10 patients who underwent pancreaticoduodenectomy (PD) for secondary periampullary cancer following extrahepatic bileduct cancer resection from 1995 to 2011. RESULTS The mean age of the 10 patients at the second operation was 61 years (range, 45-70 years). The primary cancers were 7 hilar cholangiocarcinomas, 2 middle common bile duct cancers, and one cystic duct cancer. The secondary cancers were 8 distal common bile duct cancers and 2 carcinomas of the ampulla of Vater. The second operations were 6 Whipple procedures and 4 pylorus-preserving pancreaticoduodenectomies. The mean interval between primary treatment and metachronous periampullary cancer was 20.6 months (range, 3.4-36.6 months). The distal resection margin after primary resection was positive for high grade dysplasia in one patient. Metachronous tumor was confirmed by periampullary pathology in all cases. Four of the 10 patients had delayed gastric emptying (n = 2) or pancreatic fistula (n = 2) after reoperation. There were no perioperative deaths. Median survival after PD was 44.6 months (range, 8.5-120.5 months). CONCLUSION Based on the postoperative survival rate, PD may provide an acceptable protocol for resection in patients with metachronous periampullary cancer after resection of the extrahepatic bile duct cancer.
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Affiliation(s)
- Dong Hun Kim
- Department of Surgery, Dankook University Hospital, Cheonan, Korea
| | - Dong Wook Choi
- Department of Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea
| | - Seong Ho Choi
- Department of Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea
| | - Jin Seok Heo
- Department of Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea
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9
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Takahashi K, Sasaki R, Oshiro Y, Fukunaga K, Oda T, Ohkohchi N. Well-differentiated endocrine carcinoma originating from the bile duct in association with a congenital choledochal cyst. Int Surg 2012; 97:315-320. [PMID: 23294072 PMCID: PMC3727271 DOI: 10.9738/cc152.1] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/24/2022] Open
Abstract
We encountered a rare case of a well-differentiated endocrine carcinoma originating from the bile duct in association with a congenital choledochal cyst (CCC). The patient is a 28-year-old woman referred to our clinic for pruritus. Laboratory data showed mild elevation of serum hepatobiliary enzymes. Computed tomography and magnetic resonance imaging demonstrated pancreatobiliary maljunction and a Todani type IV-A CCC from the inferior bile duct to the bilateral intrahepatic bile ducts. A solid tumor was detected in the middle portion of the common bile duct. Pancreatoduodenectomy and total extrahepatic bile duct resection was performed. Based on pathologic and immunohistochemical examinations, a diagnosis of well-differentiated endocrine carcinoma was made according to the World Health Organization criteria. To our knowledge, this is the third report of a neuroendocrine tumor originating from the bile duct in association with a CCC.
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Affiliation(s)
- Kazuhiro Takahashi
- Organ Transplantation Gastroenterological and Hepatobiliary Surgery, Division of Clinical Medicine, Faculty of Medicine, Graduate School of University of Tsukuba, Tsukuba, Japan
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Kobayashi S, Konishi M, Kato Y, Gotohda N, Takahashi S, Kinoshita T, Kinoshita T, Kojima M. Surgical outcomes of multicentric adenocarcinomas of the biliary tract. Jpn J Clin Oncol 2011; 41:1079-85. [PMID: 21875937 DOI: 10.1093/jjco/hyr103] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/27/2022] Open
Abstract
OBJECTIVE In comparison to single biliary cancers, distinct features of biliary multicentric adenocarcinomas are not yet clear. METHODS From July 1992 to July 2009, 393 patients underwent surgery for cancers of the biliary tract at the National Cancer Center Hospital East, Kashiwa, Japan. Clinicopathological characteristics and surgical outcomes of multicentric biliary adenocarcinoma were compared with those of single cancers. RESULTS During the period, 10 cases (2.5%) with multicentric cancer (6 synchronous and 4 metachronous cancers) were found among 393 cases of biliary cancer. Pathologically, compared with single cancers, multicentric adenocarcinomas were more likely to be early cancers and to be papillary carcinomas with both superficial epithelial tumor spread and extensive dysplastic epithelium, but were less likely to have lymph node metastases (P < 0.01). The proportion of multicentric cancers among early papillary cancers was high (9/24, 37.5%). Clinically, no recurrences were detected in lymph nodes, peritoneum or distant organs, but one recurrence in the remnant bile duct. Only one patient died from cancer progression. The overall survival of patients with multicentric adenocarcinomas was statistically the same as that of single cancers (median survival: 69 vs. 30 months, P = 0.47). CONCLUSIONS Multicentric adenocarcinomas of the biliary tract have distinct features compared with single cancers.
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Affiliation(s)
- Shin Kobayashi
- Department of Surgical Oncology, National Cancer Center Hospital East, Kashiwa, Chiba, Japan
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Kawai R, Miyata K, Yuasa N, Takeuchi E, Goto Y, Miyake H, Nagai H, Kobayashi Y, Ito M, Fujino M. A Case of Adenosquamous Carcinoma of the Extrahepatic Bile Duct Associated with Bipolar Differentiation in the Superficial Spreading Lesion. THE JAPANESE JOURNAL OF GASTROENTEROLOGICAL SURGERY 2011; 44:549-555. [DOI: 10.5833/jjgs.44.549] [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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Jang JY, Ahn YJ, Chung JK, Jung IM, Heo SC, Hwang KT, Ahn HS, Shin E, Seo JH. Adenosquamous Carcinoma Arising in Congenital Choledochal Cyst. JOURNAL OF THE KOREAN SURGICAL SOCIETY 2010. [DOI: 10.4174/jkss.2010.78.5.325] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/30/2022]
Affiliation(s)
- Jae Yool Jang
- Department of Surgery, Seoul National University Boramae Hospital, Seoul, Korea
| | - Young Joon Ahn
- Department of Surgery, Seoul National University Boramae Hospital, Seoul, Korea
| | - Jung Kee Chung
- Department of Surgery, Seoul National University Boramae Hospital, Seoul, Korea
| | - In Mok Jung
- Department of Surgery, Seoul National University Boramae Hospital, Seoul, Korea
| | - Seung Chul Heo
- Department of Surgery, Seoul National University Boramae Hospital, Seoul, Korea
| | - Ki-Tae Hwang
- Department of Surgery, Seoul National University Boramae Hospital, Seoul, Korea
| | - Hye Seong Ahn
- Department of Surgery, Seoul National University Boramae Hospital, Seoul, Korea
| | - Eun Shin
- Department of Pathology, Seoul National University Hospital, Seoul, Korea
| | - Ja Hee Seo
- Department of Pathology, Seoul National University Hospital, Seoul, Korea
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Terada T. Adenosquamous carcinoma in a congenital choledochal cyst associated with pancreatico-biliary maljunction. Pathol Int 2009; 59:482-5. [PMID: 19563412 DOI: 10.1111/j.1440-1827.2009.02397.x] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022]
Abstract
Congenital choledochal cyst is occasionally complicated by carcinomatous transformation, mostly adenocarcinoma. Adenosquamous carcinoma arising in a congenital choledochal cyst is very rare. The author herein reports an adenosquamous carcinoma arising in congenital choledochal cyst associated with pancreatico-biliary maljunction. A 34-year-old man with congenital choledochal cyst and recurrent cholangitis had been followed up, and was admitted to hospital to undergo testing for cancer. Imaging modalities including computed tomography, magnetic resonance imaging and endoscopic retrograde cholangiography showed an elevated lesion in the choledochal cyst. Because clinical cytology of bile indicated malignant cells, pancreatico-duodenectomy, cholecystectomy, and resection of the choledochal cyst were performed. Grossly, the choledochal cyst was type I, and its size was 8 x 10 cm. Anomalous pancreatico-biliary ductal union was recognized. An elevated lesion was recognized in the choledochal cyst. Histologically, the lesion was composed of a squamous cell carcinoma element and an adenocarcinoma element; a gradual transition was recognized between the two. The squamous cell carcinoma element contained microcytic cells with mucins. On immunohistochemistry the adenocarcinoma element and microcytic cells were positive for CEA, but the squamous cell carcinoma element was negative for CEA. Both elements were positive for CA19-9. Ki-67 labeling was 53% in the adenocarcinoma element and 48% in the squamous cell carcinoma element. p53 protein was negative in both elements. At the time of writing, the patient was alive after 25 months without recurrence or metastasis. The present case is the second case of adenosquamous carcinoma arising in congenital choledochal cyst in the English-language literature.
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Affiliation(s)
- Tadashi Terada
- Department of Pathology, Shizuoka City Shimizu Hospital, Shizuoka, Japan.
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14
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Pancreaticobiliary maljunction and carcinogenesis to biliary and pancreatic malignancy. Langenbecks Arch Surg 2008; 394:159-69. [PMID: 18500533 DOI: 10.1007/s00423-008-0336-0] [Citation(s) in RCA: 110] [Impact Index Per Article: 6.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/04/2008] [Accepted: 04/04/2008] [Indexed: 02/07/2023]
Abstract
BACKGROUND It is widely accepted that congenital choledochal cyst is associated with pancreaticobiliary maljunction (PBM). But, PBM is an independent disease entity from choledochal cyst. PBM is synonymous with "abnormal junction of the pancreaticobiliary ductal system", "anomalous arrangement of pancreaticobiliary ducts", "anomalous union of bilio-pancreatic ducts", etc. Cases with PBM not associated with biliary duct dilatation are often found, and these cases are frequently complicated gallbladder cancer. The Japanese Study Group of Pancreaticobiliary Maljunction was started in 1983, and defined diagnostic criteria and nationwide registration system of PBM cases was started. PBM is defined as a union of the pancreatic and biliary ducts which is located outside the duodenal wall. Bile and pancreatic juice reflux and regurgitate mutually. BILIARY CARCINOGENESIS The most bothersome problem is biliary carcinogenesis. Gallbladder cancers arise in 14.8% and bile duct cancers arise in 4.9%. The incidence of the gallbladder carcinoma of PBM without bile duct dilatation is 36.1%. Many investigators have tried to clarify the carcinogenic process, from various aspects. The biliary epithelia are injured by harmful substances, and in the course of repair, multiple alterations of oncogenes and tumor suppressor genes are followed, and they lead to carcinoma through multistage interaction. In the biliary epithelia of PBM, incidence and degree of hyperplasia are characteristic. K-ras gene mutations are observed in the cancerous as well as noncancerous lesions of biliary tract of PBM patients. Mutations of p53 gene and overexpression of p53 protein are also found in the cancerous and noncancerous lesions. These changes are called "hyperplasia-carcinoma sequence". TREATMENT Total excision of the extrahepatic bile duct with gallbladder followed by hepaticojejunostomy, Roux-en-Y, or end-to-side hepaticoduodenostomy are treatment of choice, even for cases with not dilated bile duct, because the incidence of cancer in the nondilated bile duct is not negligible, and genetic changes are seen in a nondilated bile duct.
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Adenosquamous carcinoma of the extrahepatic bile duct: clinicopathologic and radiologic features. ACTA ACUST UNITED AC 2008; 34:217-24. [PMID: 18204917 DOI: 10.1007/s00261-008-9363-0] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/22/2022]
Abstract
BACKGROUND To retrospectively analyze computed tomography (CT) features of adenosquamous carcinoma (ASC) of the extrahepatic bile duct and to correlate them with pathologic findings. METHODS Six patients who underwent radical surgical resection for ASC of extrahepatic bile duct were included. CT and pathologic findings were analyzed and correlated with each other. RESULTS Five ASCs were periductal infiltrative type and the remaining one was intraductal polypoid type. Histologically, ASC was composed of squamous and glandular components which mixed in an intermingled pattern and was characterized by a high local aggressiveness mainly due to the squamous component. The most common CT findings were a segmental wall thickening with delayed enhancement. Overall, CT features were well correlated with pathologic findings. However, CT tended to underestimate radial extent of the tumor compared with histologic findings. CONCLUSIONS Histopathologically, ASC of the extrahepatic bile duct is characterized by greater local aggressiveness. However, their CT findings are not quite different from those of usual adenocarcinoma due to the unique geographic characteristic of being located at a very limited space.
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Piessen G, Mariette C, Aubry E, Triboulet JP. [Duodenal atresia and choledochal cyst: a rare combination]. GASTROENTEROLOGIE CLINIQUE ET BIOLOGIQUE 2006; 30:1085-9. [PMID: 17075456 DOI: 10.1016/s0399-8320(06)73481-9] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 05/12/2023]
Abstract
Congenital duodenal atresia is usually associated with various congenital anomalies. The embryological events in the development of the biliary tract and duodenum are linked. Although an association between duodenal atresia and biliary anomalies is predictable, it is rarely observed. We describe the case of a 27 year-old woman, operated on for duodenal atresia in her childhood, who presented cholangiolitis. Morphologic investigation allowed the diagnosis of choledochal cyst with an intracystic stone developed in the pancreatic head. A pancreatico-duodenectomy was performed because of the importance and the location of dilatation and stone with biliary obstruction. To our knowledge, the association between a congenital duodenal atresia and a choledochal cyst revealed at adult age had never been described in the literature.
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Affiliation(s)
- Guillaume Piessen
- Service de Chirurgie digestive et générale, Hôpital Claude Huriez, CHRU, Place de Verdun, 59037 Lille Cedex
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Wu GS, Zou SQ, Liu ZR, Wang DY. Bile from a patient with anomalous pancreaticobiliary ductal union promotes the proliferation of human cholangiocarcinoma cells via COX-2 pathway. World J Gastroenterol 2003; 9:1094-7. [PMID: 12717864 PMCID: PMC4611380 DOI: 10.3748/wjg.v9.i5.1094] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/06/2023] Open
Abstract
AIM: To explore the effects of COX-2 gene in the proliferative activity induced by bile from anomalous pancreaticobiliary ductal union (APBDU) on human cholangiocarcinoma cell line.
METHODS: Bile sample from APBDU and normal bile sample were used for this study. The proliferative effect of bile was measured by methabenzthiazuron (MTT) assay; COX-2 mRNA was examined by semi-quantitative reverse transcription polymerase chain reaction (RT-PCR). Cell cycle was analyzed by flow cytometry (FCM), and the PGE2 levels in the supernatant of cultured cholangiocarcinoma cells were quantitated by enzyme-linked immunoabsordent assay (ELISA).
RESULTS: Bile from APBDU can significantly promote the proliferation of human cholangiocarcinoma QBC939 cells compared with normal bile (P = 0.005) and up-regulated remarkably their COX-2 mRNA expression (P = 0.004). The proliferative activity of APBDU bile can be abolished by addition of cyclooxygenase-2 specific inhibitor celecoxib.
CONCLUSION: Bile from APBDU can promote the proliferation of human cholangiocarcinoma QBC939 cells via COX-2 pathway.
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Affiliation(s)
- Gao-Song Wu
- Department of General Surgery, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, 430030, Hubei Province, China.
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Okamoto A, Tsuruta K, Matsumoto G, Takahashi T, Kamisawa T, Egawa N, Funata N. Papillary carcinoma of the extrahepatic bile duct: characteristic features and implications in surgical treatment. J Am Coll Surg 2003; 196:394-401. [PMID: 12648691 DOI: 10.1016/s1072-7515(02)01664-2] [Citation(s) in RCA: 20] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/07/2023]
Abstract
BACKGROUND Papillary carcinoma of the extrahepatic bile duct presents clinically and histologically distinct features relevant to surgical decision-making. STUDY DESIGN Serial sections of 15 specimens of resected papillary carcinoma of the bile duct were histologically examined to determine mode of spread, possibility of multicentric tumor origins, and coincidence with other neoplastic lesions. The presence of anomalous pancreaticobiliary ductal union was also investigated. These characteristics were considered with regard to surgical treatment. RESULTS Three patients displaying pancreaticobiliary maljunction and one of three patients with a long common channel (> or = 8 mm) exhibited multicentric tumors. Eight patients (53%) demonstrated superficial spread along a mean length of 37.8 mm (range, 5 to 67 mm) of bile duct mucosa. Multicentric tumors developed synchronously in 4 patients, while metachronous tumors were identified in three patients displaying tumor histology similar to the primary lesions. Two of these three underwent successful repeated resection. Concomitant neoplastic lesions in the biliary tract were identified as mucosal dysplasia in four patients and cholangiocellular carcinoma of the liver in two. All tumors but one were removed via hepatic lobectomy or pancreatoduodenectomy, or both, resulting in a 5-year survival rate of 60%. CONCLUSIONS Aggressive resection offers clear survival benefits for patients presenting with tumors displaying extensive superficial spread or multicentric origins. Closer attention should be paid to long common channels in relation to carcinogenesis of the bile duct, in addition to pancreaticobiliary maljunction. The risk of secondary tumor development remains, particularly in patients with pancreaticobiliary maljunction even after excision of the tumor-bearing extrahepatic bile duct.
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Affiliation(s)
- Atsutake Okamoto
- Department of Surgery, Tokyo Metropolitan Komagome Hospital, Japan
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