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Hayasaki A, Tanemura A, Uchida K, Nagata M, Yamada R, Fujii T, Murata Y, Kuriyama N, Kishiwada M, Mizuno S. Choledochocele with hyperplastic epithelium in a patient who developed severe acute pancreatitis and underwent subtotal stomach-preserving pancreatoduodenectomy: a case report. Clin J Gastroenterol 2024; 17:170-176. [PMID: 37815654 DOI: 10.1007/s12328-023-01870-2] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/10/2023] [Accepted: 09/22/2023] [Indexed: 10/11/2023]
Abstract
Choledochocele is defined as a congenital dilatation of the distal intramural part of the common bile duct protruding into the wall of the descending duodenum, typically without pancreaticobiliary maljunction. However, some cases present with a similar pathophysiology to pancreaticobiliary maljunction, including reciprocal reflux of pancreatic juices and bile, leading to protein plugs, pancreatitis, and biliary tract carcinogenesis. Choledochocele is relatively rare and its anatomy, physiology, pathology, and clinical features are thus not well known. We describe a patient with choledochocele who suffered from repeated severe acute pancreatitis and underwent subtotal stomach-preserving pancreatoduodenectomy, in whom the pathological findings of choledochocele showed hyperplasia.
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Affiliation(s)
- Aoi Hayasaki
- Department of Hepato-Biliary-Pancreatic and Transplant Surgery, School of Medicine, Mie University, 2-174 Edobashi, Tsu, Mie, 514-8507, Japan.
| | - Akihiro Tanemura
- Department of Hepato-Biliary-Pancreatic and Transplant Surgery, School of Medicine, Mie University, 2-174 Edobashi, Tsu, Mie, 514-8507, Japan
| | - Katsunori Uchida
- Department of Oncologic Pathology, Mie University, Tsu, Mie, Japan
| | | | - Reiko Yamada
- Department of Gastroenterology and Hepatology, Mie University, Tsu, Mie, Japan
| | - Takehiro Fujii
- Department of Hepato-Biliary-Pancreatic and Transplant Surgery, School of Medicine, Mie University, 2-174 Edobashi, Tsu, Mie, 514-8507, Japan
| | - Yasuhiro Murata
- Department of Hepato-Biliary-Pancreatic and Transplant Surgery, School of Medicine, Mie University, 2-174 Edobashi, Tsu, Mie, 514-8507, Japan
| | - Naohisa Kuriyama
- Department of Hepato-Biliary-Pancreatic and Transplant Surgery, School of Medicine, Mie University, 2-174 Edobashi, Tsu, Mie, 514-8507, Japan
| | - Masashi Kishiwada
- Department of Hepato-Biliary-Pancreatic and Transplant Surgery, School of Medicine, Mie University, 2-174 Edobashi, Tsu, Mie, 514-8507, Japan
| | - Shugo Mizuno
- Department of Hepato-Biliary-Pancreatic and Transplant Surgery, School of Medicine, Mie University, 2-174 Edobashi, Tsu, Mie, 514-8507, Japan
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Shrateh ON, khader A, Musleh A, Abbadi K, Asbah M, Sawalha N, Jobran AW. Successful radiological detection and surgical management of type 3 choledochocele: A case report. Radiol Case Rep 2023; 18:1502-1506. [PMID: 36816336 PMCID: PMC9932293 DOI: 10.1016/j.radcr.2023.01.029] [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: 12/29/2022] [Revised: 01/05/2023] [Accepted: 01/07/2023] [Indexed: 02/05/2023] Open
Abstract
According to Todani's classification, a choledochocele is a cystic dilatation of the distal section of the common bile duct that protrudes into the duodenal lumen. It is also known as a type III choledochal cyst. There are not many cases, and the cause is still unknown. Typically, it is misdiagnosed as a peptic ulcer, as was the case with the patient in this case study. For choledochocele diagnosis, multislice spiral computed tomography and magnetic resonance cholangiopancreatography may be equivalent to endoscopic retrograde cholangiography. Both endoscopic therapy and open surgical care are risk-free possibilities, and the cyst's size influences which strategy should be used. A 35-year-old woman admitted to the hospital with upper abdominal pain caused by a large choledochocele was successfully treated by open surgical management. In this case study, we outline the specifics of her situation and talk about the most recent research on similar cases and how to treat them therapeutically. There is ongoing debate regarding the best course of action. However, to achieve a successful outcome, open surgical care for choledochocele of large size (ie, > 3 cm in diameter) is advised.
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Affiliation(s)
- Oadi N. Shrateh
- Medical Research Club, Faculty of Medicine, Al-Quds University, Jerusalem, Palestine
- Corresponding author.
| | - Abdullatif khader
- Faculty of Medicine, Al-Quds University, Jerusalem, Palestine
- General Surgery Department, Palestinian Medical Complex, Ramallah, Palestine
| | - Asil Musleh
- Department of General Surgery, Palestinian Medical Complex, Ramallah, Palestine
| | - Khaled Abbadi
- Department of General Surgery, Palestinian Medical Complex, Ramallah, Palestine
| | - Malvina Asbah
- Department of General Surgery, Palestinian Medical Complex, Ramallah, Palestine
| | - Nihal Sawalha
- Faculty of Medicine, Al-Quds University, Jerusalem, Palestine
- Department of General Surgery, Palestinian Medical Complex, Ramallah, Palestine
| | - Afnan W.M. Jobran
- Medical Research Club, Faculty of Medicine, Al-Quds University, Jerusalem, Palestine
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Bulotta AL, Stern MV, Moneghini D, Parolini F, Bondioni MP, Missale G, Boroni G, Alberti D. Endoscopic treatment of periampullary duodenal duplication cysts in children: Four case reports and review of the literature. World J Gastrointest Endosc 2021; 13:529-542. [PMID: 34733413 PMCID: PMC8546566 DOI: 10.4253/wjge.v13.i10.529] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/28/2021] [Revised: 04/30/2021] [Accepted: 08/02/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Duodenal duplications are rare congenital anomalies of the gastrointestinal tract. As the periampullary variant is much rarer, literature is scant and only few authors have reported their experience in diagnosis and treatment, particularly with operative endoscopy.
CASE SUMARY To report our experience with the endoscopic treatment in a series of children with periampullary duodenal duplication cysts, focusing on the importance of obtaining an accurate preoperative anatomic assessment of the malformations. The pediatric periampullary duodenal duplication cyst literature is reviewed. We conducted a systematic review according to the PRISMA guidelines. The PubMed database was searched for original studies on “duodenal duplication”, “periampullary duplication” or “endoscopic management” published since 1990, involving patients younger than 18 years of age. Eligible study designs were case report, case series and reviews. We analyzed the data and reported the results in table and text. Fifteen eligible articles met the inclusion criteria with 16 patients, and analysis was extended to our additional 4 cases. Median age at diagnosis was 13.5 years. Endoscopic treatment was performed in 10 (50%) patients, with only 2 registered complications.
CONCLUSION Periampullary duodenal duplication cysts in pediatric patients are very rare. Our experience suggests that an accurate preoperative assessment is critical. In the presence of sludge or stones inside the duplication, endoscopic retrograde cholangio-pancreatography is mandatory to demonstrate a communication with the biliary tree. Endoscopic treatment resulted in a safe, minimally invasive and effective treatment. In periampullary duodenal duplication cyst endoscopically treated children, long-term follow-up is still necessary considering the potential malignant transformation at the duplication site.
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Affiliation(s)
- Anna Lavinia Bulotta
- Department of Pediatric Surgery, Azienda Socio Sanitaria Territoriale degli Spedali Civili di Brescia, Brescia 25123, Italy
| | - Maria Vittoria Stern
- Department of Pediatric Surgery, Azienda Socio Sanitaria Territoriale degli Spedali Civili di Brescia, Brescia 25123, Italy
| | - Dario Moneghini
- Department of Digestive Endoscopy, Azienda Socio Sanitaria Territoriale degli Spedali Civili di Brescia, Brescia 25123, Italy
| | - Filippo Parolini
- Department of Pediatric Surgery, Azienda Socio Sanitaria Territoriale degli Spedali Civili di Brescia, Brescia 25123, Italy
| | - Maria Pia Bondioni
- Department of Pediatric Radiology, Azienda Socio Sanitaria Territoriale degli Spedali Civili di Brescia, Brescia 25123, Italy
| | - Guido Missale
- Department of Digestive Endoscopy, Azienda Socio Sanitaria Territoriale degli Spedali Civili di Brescia, Brescia 25123, Italy
| | - Giovanni Boroni
- Department of Pediatric Surgery, Azienda Socio Sanitaria Territoriale degli Spedali Civili di Brescia, Brescia 25123, Italy
| | - Daniele Alberti
- Department of Pediatric Surgery, Azienda Socio Sanitaria Territoriale degli Spedali Civili di Brescia, Brescia 25123, Italy
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Aydin Mericöz C, Hacihasanoglu E, Muraki T, Pehlivanoglu B, Memis B, Mittal P, Polito H, Saka B, Everett R, Sarmiento J, Kooby D, Maithel SK, Erkan M, Basturk O, Reid MD, Adsay V. Evaluation and Pathologic Classification of Choledochal Cysts: Clinicopathologic Analysis of 84 Cases From the West. Am J Surg Pathol 2021; 45:627-637. [PMID: 33481384 DOI: 10.1097/pas.0000000000001666] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/07/2023]
Abstract
Choledochal cyst (CC) is believed to be a mostly Asian disorder. As a clinically defined entity, its pathologic correlates are poorly characterized. Eighty-four resected CCs from the West were reanalyzed. After applying established Japanese criteria, 9/66 with available imaging were disqualified and 10/39 with preoperative cyst typing had to be recategorized. None had been diagnosed with, or evaluated for, pancreatobiliary maljunction, but on retrospective analysis of radiologic images, 12/66 were found to have pancreatobiliary maljunction. The clinical findings were: F/M=5.7; mean age, 48; most (77%) presented with abdominal pain; mean size, 2.9 cm; choledocholithiasis 11%. Gross/histologic examination revealed 3 distinct pathology-based categories: (I) Cystic dilatation of native ducts (81%). (II) Double bile duct (13%), almost all of which were found in women (10/11); all were diagnosed by pathologic examination, and not preoperative diagnosis. (III) Gastrointestinal (GI) duplication type (6%). Microscopic findings of the entire cohort included mucosal-predominant lymphoplasmacytic inflammation (50%), follicular cholangitis (7%), mucosal hyperplasia (43%; 13% with papillae), intestinal metaplasia (10%), BilIN-like hyperplasia (17%), erosion/ulceration (13%), and severe dysplasia-mimicking atypia including "detachment atypia" and micropapillary degeneration (11%). Carcinomatous changes were seen in 14 cases (17%) (high-grade dysplasia/carcinoma in situ in 7, intraductal papillary neoplasm 1, and invasive carcinoma 6); and 13/14 of these occurred in pathologic category I, all with cyst size >1 cm. In conclusion, diagnostic imaging guidelines used in Asia are not routinely used (but should be adopted) in the West. Pathologically, cases designated as CC are classifiable in 3 groups: category 1 (dilated native duct type), more prone to carcinomatous change; category 2, double-duct phenomenon (all but 1 being female in this study); and category 3, GI-type duplication. Overall, 17% of CCs show carcinomatous change (50% of them invasive). CC specimens should be carefully examined with this classification and submitted entirely for assessment of at-risk mucosa and cancerous transformation.
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Affiliation(s)
| | | | | | | | | | | | | | | | | | | | | | | | - Mert Erkan
- Surgery, Koç University School of Medicine
- Koç University Research Center for Translational Medicine (KUTTAM), Istanbul, Turkey
| | - Olca Basturk
- Department of Pathology, Memorial Sloan Kettering Cancer Center, New York, NY
| | | | - Volkan Adsay
- Departments of Pathology
- Koç University Research Center for Translational Medicine (KUTTAM), Istanbul, Turkey
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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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Yang J, Xiao GF, Li YX. Open surgical treatment of choledochocele: A case report and review of literature. World J Clin Cases 2018; 6:842-846. [PMID: 30510953 PMCID: PMC6264990 DOI: 10.12998/wjcc.v6.i14.842] [Citation(s) in RCA: 5] [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: 09/09/2018] [Revised: 10/16/2018] [Accepted: 10/23/2018] [Indexed: 02/05/2023] Open
Abstract
Choledochocele (also known as type III choledochal cyst according to Todani’s classification) is a cystic dilation of the distal segment of the common bile duct protruding into the duodenal lumen. Cases are rare and the etiology remains unclear. It is usually misdiagnosed as peptic ulcer, as in the patient whose case is described here. Multislice spiral computed tomography and magnetic resonance cholangiopancreatography may be comparable to endoscopic retrograde cholangiography for diagnosis of choledochocele. Both endoscopic therapy and open surgical management are safe options, and size of the cyst plays a role in the decision-making for which approach to apply. A 50-year-old woman admitted to our hospital with upper abdominal pain caused by choledochocele with large size was successfully treated by open surgical management. We present the details of her case in this case report and discuss the recent literature on such cases and their therapeutic management.
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Affiliation(s)
- Jie Yang
- Department of Biliopancreatic Surgery, Xiangya Hospital, Central South University, Changsha 410008, Hunan Province, China
| | - Guang-Fa Xiao
- Department of Biliopancreatic Surgery, Xiangya Hospital, Central South University, Changsha 410008, Hunan Province, China
| | - Yi-Xiong Li
- Department of Biliopancreatic Surgery, Xiangya Hospital, Central South University, Changsha 410008, Hunan Province, China
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Lobeck IN, Dupree P, Falcone RA, Lin TK, Trout AT, Nathan JD, Tiao GM. The presentation and management of choledochocele (type III choledochal cyst): A 40-year systematic review of the literature. J Pediatr Surg 2017; 52:644-649. [PMID: 27829523 DOI: 10.1016/j.jpedsurg.2016.10.008] [Citation(s) in RCA: 16] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/13/2016] [Revised: 08/24/2016] [Accepted: 10/11/2016] [Indexed: 12/11/2022]
Abstract
BACKGROUND Choledochoceles may cause biliary obstruction and harbor malignancy. We conducted a 40-year systematic review of the literature for this rare anomaly. METHODS PubMed and Cochrane databases were accessed 1975-2015 using terms "choledochocele" or "choledochal cyst". Studies reviewed that met the following criteria: English language, published 1975-2015 with human subjects. RESULTS 325 patients with a choledochocele were identified, including 71 case reports and 254 cases within institutional reviews. 13 pediatric case reports of choledochocele exist, with abdominal pain being the most common symptom (n=11). The most frequent diagnostic and treatment modalities were ultrasound (n=10), and endoscopic sphincterotomy (n=5). No malignancies were reported. 58 adult case reports exist, with the most common presenting symptom being abdominal pain (n=54). Ultrasound was the frequently employed diagnostic modality (n=32). Open procedures were performed more often (n=30). Malignant lesions were identified in 5. In 42 institutional reviews, the frequency of choledochocele was 0.7%. Of those for whom treatment was reported, 69% underwent endoscopic sphincterotomy. CONCLUSION Choledochocele is a rare malformation. Similarities exist between pediatric and adult patients, but malignancy has only been reported in adults. An algorithm based on patient age, cyst size, lining and amenability to endoscopic resection may be considered as a treatment strategy for this uncommon condition.
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Affiliation(s)
- Inna N Lobeck
- Division of Pediatric General and Thoracic Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States
| | - Phylicia Dupree
- Division of Pediatric General and Thoracic Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States
| | - Richard A Falcone
- Division of Pediatric General and Thoracic Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States
| | - Tom K Lin
- Division of Gastroenterology, Hepatology & Nutrition, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States
| | - Andrew T Trout
- Department of Radiology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States
| | - Jaimie D Nathan
- Division of Pediatric General and Thoracic Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States
| | - Greg M Tiao
- Division of Pediatric General and Thoracic Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States.
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9
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Lobeck IN, Dupree P, Falcone RA, Lin TK, Trout AT, Nathan JD, Tiao GM. Transduodenal resection of a choledochocele (type III choledochal cyst) with sphincteroplasty: A case report. JOURNAL OF PEDIATRIC SURGERY CASE REPORTS 2016. [DOI: 10.1016/j.epsc.2016.04.007] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/26/2022] Open
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Zhu L, Lv Z, Liu J, Xu W. Choledochocele: A Case Report and Discussion of Diagnosis Criteria. European J Pediatr Surg Rep 2015; 3:85-9. [PMID: 26788455 PMCID: PMC4712052 DOI: 10.1055/s-0035-1563601] [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: 03/26/2015] [Accepted: 07/03/2015] [Indexed: 02/06/2023] Open
Abstract
A 6-year-old girl presented with intermittent abdominal pain, without jaundice and a palpable mass in the epigastrium. Preoperative imaging and upper endoscopy suggested duodenal duplication. During surgery, the patient was diagnosed with a rare type of choledochal cyst—choledochocele (type 3b). The authors emphasize that, in children, choledochocele should be included in the differential diagnosis of cystic lesions located in the duodenal area and the head of the pancreas area, regardless of jaundice or abnormal liver function. Since mucosal histology showing duodenal mucosa did not match the final diagnosis, we suggest that three criteria should be met for the diagnosis of a choledochocele to be diagnosed: (1) a cyst protruding into the duodenal lumen; (2) filling with contrast during cholangiography and (3) a filling defect on X-ray barium meal.
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Affiliation(s)
- Linlin Zhu
- Department of Surgery, Children's Hospital of Shanghai Jiaotong University, Shanghai, China
| | - Zhibao Lv
- Department of Surgery, Children's Hospital of Shanghai Jiaotong University, Shanghai, China
| | - Jiangbin Liu
- Department of Surgery, Children's Hospital of Shanghai Jiaotong University, Shanghai, China
| | - Weijue Xu
- Department of Surgery, Children's Hospital of Shanghai Jiaotong University, Shanghai, China
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Kwak TY, Park CH, Eom SH, Hwang HS, Chung DW, Seo JY, Kim YS, Kwak DH. Choledochocele containing a stone mistaken as a distal common bile duct stone. Yeungnam Univ J Med 2015. [DOI: 10.12701/yujm.2015.32.1.60] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022] Open
Affiliation(s)
- Tae Young Kwak
- Department of Internal Medicine, Kwak's Hospital, Daegu, Korea
| | - Chang Hwan Park
- Department of Internal Medicine, Kwak's Hospital, Daegu, Korea
| | - Seok Hyeon Eom
- Department of Internal Medicine, Kwak's Hospital, Daegu, Korea
| | - Hong Suk Hwang
- Department of Internal Medicine, Kwak's Hospital, Daegu, Korea
| | - Duk Won Chung
- Department of Internal Medicine, Kwak's Hospital, Daegu, Korea
| | - Ji Young Seo
- Department of Internal Medicine, Kwak's Hospital, Daegu, Korea
| | - Yeong Sung Kim
- Department of Internal Medicine, Kwak's Hospital, Daegu, Korea
| | - Dong Hyup Kwak
- Department of Internal Medicine, Kwak's Hospital, Daegu, Korea
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Law R, Topazian M. Diagnosis and treatment of choledochoceles. Clin Gastroenterol Hepatol 2014; 12:196-203. [PMID: 23660418 DOI: 10.1016/j.cgh.2013.04.037] [Citation(s) in RCA: 46] [Impact Index Per Article: 4.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/11/2013] [Revised: 04/15/2013] [Accepted: 04/16/2013] [Indexed: 02/07/2023]
Abstract
Choledochoceles are cystic dilatations of the intraduodenal portion of the common bile duct. Although often classified as Type III biliary cysts, choledochoceles have distinctive demographic and anatomic features and a lower risk of malignancy than other types of choledochal cysts. Type A choledochoceles are cystic dilatations of a segment of the intra-ampullary bile duct and are located proximal to the ampullary orifice. Type B choledochoceles are diverticula of the intra-ampullary common channel and are located distal to the ampullary orifice; they can be distinguished from duodenal duplication cysts both anatomically and histologically. Both types of choledochocele may present with pancreatitis, biliary obstruction, or nonspecific gastrointestinal symptoms. Cross-sectional imaging, endoscopic ultrasound, and endoscopic retrograde cholangiopancreatography are useful for diagnosis. Choledochoceles may be drained or resected endoscopically. Surveillance for dysplasia should be considered for lesions that are not resected.
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Affiliation(s)
- Ryan Law
- Division of Gastroenterology and Hepatology, Mayo Clinic, Rochester, Minnesota
| | - Mark Topazian
- Division of Gastroenterology and Hepatology, Mayo Clinic, Rochester, Minnesota.
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Ando H, Takada T. Cystic Disorders of the Bile Ducts. SHACKELFORD'S SURGERY OF THE ALIMENTARY TRACT 2013:1397-1404. [DOI: 10.1016/b978-1-4377-2206-2.00111-1] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/03/2025]
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Abstract
The classification of choledochoceles as a type of choledochal cyst stems from the 1959 article by Alonso-Lej and colleagues describing 94 choledochal cysts, only 4 of which were choledochoceles. Even then, Alonso-Lej questioned the propriety of including the choledochocele, stating it was unclear "as to whether or not it originates from the same etiologic factors [as other choledochal cysts]". In 1971, Trout and Longmire also questioned the validity of classifying choledochoceles as choledochal cysts, noting the anatomic position article and variant mucosa of the choledochocele. Wearn and Wiot, in an article titled "Choledochocele: not a form of choledochal cyst", cite the differences in clinical presentation, demographics, and histology as reasons why choledochoceles represent separate entities from choledochal cysts. Over the ensuing decades, numerous investigators have questioned the legitimacy of classifying choledochoceles as choledochal cysts. In our recent series (the only one to our knowledge directly comparing patients with choledochocele and other [type I, II, IV, and V] choledochal cysts), patients with choledochoceles differed from patients with choledochal cysts in their age, gender, presenting symptoms, history of previous cholecystectomy, pancreatobiliary ductal anatomy, management, and most importantly, propensity to developing biliary malignancy. Based on the available cases of choledochoceles found in the literature, combined with the recent series from our institution, we conclude that choledochoceles seem to be distinct entities from choledochal cysts.
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Affiliation(s)
- Kathryn M Ziegler
- Department of Surgery, Indiana University, 545 Barnhill Drive, EH 202, Indianapolis, IN 46202, USA
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Periampullary choledochal diverticula are not choledochal cysts. Gastrointest Endosc 2011; 73:994-1001. [PMID: 21439566 DOI: 10.1016/j.gie.2011.01.013] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/23/2010] [Accepted: 01/04/2011] [Indexed: 02/08/2023]
Abstract
BACKGROUND Choledochal cysts (CC) are rare, congenital anomalies of the biliary tree, associated with the development of biliary malignancies. Small periampullary choledochal diverticula (PCD) are a previously unreported type of biliary anomaly found primarily at ERCP. OBJECTIVE The aim of this study was to assess whether PCD are congenital or acquired lesions by comparing the clinical presentation, management, and risk of malignancy between patients with PCD and CC. DESIGN Retrospective analysis of a medical center database. SETTING Academic tertiary referral center. PATIENTS Over the study period, data regarding 16 patients with PCD were identified and compared with that of 118 patients with CC. INTERVENTION Retrospective review of ERCP, surgical pathology, billings, and a diagnostic imaging database from our institution from 1985 to 2009 was done. MAIN OUTCOME MEASUREMENTS Clinical presentation, investigations, management strategies, complication rates, and long-term outcomes were compared in patients with classic CC and PCD over the same time period. RESULTS Patients with PCD were less likely to be female (50% vs 81%), older aged (mean 68 vs 28 years), to complain of abdominal pain (88% vs 68%), and were less likely to present with jaundice (0% vs 32%) (P<.05 for all pairs). Patients with PCD also were noted to have lower frequency of anomalous pancreatobiliary junction (0% vs 83%) and biliary neoplasia (0% vs 5%) and more likely to have sphincter of Oddi dysfunction (63% vs 1%). Management of PCD was done with ERCP in 87% of cases and with surgery in 0% of cases, whereas management of CC was done with ERCP in 20% of cases and surgery in 80% of cases (P<.001). Long-term complications at a mean follow-up of 3.7 years after therapy were more common in CC (40% vs 6%, P=.02). LIMITATIONS Retrospective study. Lack of structured follow up. CONCLUSION Small, periampullary, choledochal diverticula are a newly reported, likely acquired anomaly of the biliary tract that are frequently associated with sphincter of Oddi dysfunction and may be secondary to biliary hypertension. These acquired lesions should not be classified as CC.
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Abstract
OBJECTIVE The aim of this analysis was to report a multidisciplinary series comparing choledochoceles to Todani Types I, II, IV, and V choledochal cysts. SUMMARY BACKGROUND DATA Choledochoceles have been classified as Todani Type III choledochal cysts. However, most surgical series of choledochal cysts have reported few choledochoceles because they are managed primarily by endoscopists. METHODS Surgical, endoscopic, and radiologic records were reviewed at the Riley Children's Hospital and the Indiana University Hospitals to identify patients with choledochal cysts. Patient demographics, presenting symptoms, radiologic studies, associated abnormalities, surgical and endoscopic procedures as well as outcomes were reviewed. RESULTS A total of 146 patients with "choledochal cysts" including 45 children (31%) and 28 with choledochoceles (18%) were identified, which represents the largest Western series. Patients with choledochoceles were older (50.7 vs. 29.0 years, P < 0.05) and more likely to be male (43% vs. 19%, P < 0.05), to present with pancreatitis (48% vs. 24%, P < 0.05) rather than jaundice (11% vs. 30%, P < 0.05) or cholangitis (0% vs. 21%, P < 0.05), to have pancreas divisum (38% vs. 10%, P < 0.01), and to be managed with endoscopic therapy (79% vs. 17%, P < 0.01). Two patients with choledochoceles (7%) had pancreatic neoplasms. CONCLUSIONS Patients with choledochoceles differ from patients with choledochal cysts with respect to age, gender, presentation, pancreatic ductal anatomy, and their management. The association between choledochoceles and pancreas divisum is a new observation. Therefore, we conclude that classifications of choledochal cysts should not include choledochoceles.
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Urushihara N, Fukumoto K, Fukuzawa H, Suzuki K, Matsuoka T, Kawashima S, Watanabe K, Hasegawa S. Recurrent pancreatitis caused by pancreatobiliary anomalies in children with annular pancreas. J Pediatr Surg 2010; 45:741-6. [PMID: 20385281 DOI: 10.1016/j.jpedsurg.2009.05.027] [Citation(s) in RCA: 15] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/20/2009] [Revised: 05/18/2009] [Accepted: 05/19/2009] [Indexed: 10/24/2022]
Abstract
PURPOSE Annular pancreas (AP) is usually associated with duodenal obstruction in neonates. Pancreatitis with AP occurs frequently in adults but is rare in children. This article describes pancreatitis in children with AP and pancreatobiliary anomalies and its surgical treatment. PATIENTS AND METHODS Six children who underwent duodenal bypass for AP subsequently developed recurrent pancreatitis. Three had trisomy 21. Duodenoduodenostomy had been performed in 5 patients and gastrojejunostomy in 1 patient for neonatal duodenal obstruction. We reviewed overall management, imaging, and surgical treatment in these children. RESULTS All children subsequently complained of recurrent abdominal pain. Pancreatitis developed in 6 children, and magnetic resonance cholangiopancreatography and endoscopic retrograde cholangiopancreatography (ERCP) revealed associated pancreatobiliary anomalies such as pancreas divisum, pancreatobiliary malunion, choledochocele, and intraluminal duodenal diverticulum. In 5 cases, surgery for recurrent or chronic pancreatitis was performed. The range of follow-up was 11 to 54 months, and all children who underwent surgery had excellent results. CONCLUSIONS Children with AP occasionally require reoperation for recurrent pancreatitis because of associate pancreatobiliary anomalies. Magnetic resonance cholangiopancreatography and ERCP provide excellent images of pancreatobiliary anomalies. Intraoperative cholangiopancreatography is also essential for accurate depiction of the ductal structure and selection of the appropriate surgical procedure.
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Affiliation(s)
- Naoto Urushihara
- Department of Pediatric Surgery, Shizuoka Children's Hospital, 860 Urushiyama, Aoi-ku, Shizuoka 420-8660, Japan.
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18
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Sonoda M, Sato M, Miyauchi Y, Yazumi S, Nakamura M. A rare case of choledochocele associated with pancreas divisum. Pediatr Surg Int 2009; 25:991-4. [PMID: 19690869 DOI: 10.1007/s00383-009-2460-5] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/07/2023]
Abstract
Choledochocele, type III choledochal cyst, is a rare abnormality. We report a rare case of choledochocele involving a 15-year-old girl who presented with intermittent episodes of upper abdominal pain. She underwent computed tomography (CT) and magnetic resonance cholangiopancreatography (MRCP), which revealed no abnormalities of biliary and pancreatic ducts. Endoscopic retrograde cholangiopancreatography (ERCP) initially suggested a diagnosis of pancreas divisum (PD), and so we failed to diagnose choledochocele at that time. Her condition improved and she was discharged, but she presented again with recurrent episodes of upper abdominal pain. Then, spiral computed tomography with intravenous infusion cholangiography (IVC-SCT) facilitated a diagnosis of choledochocele. The coexistence of choledochocele and pancreas divisum is extremely rare, and it is difficult to diagnose. However, this case suggests that the absence of pancreatitis does not rule out choledochal cysts, and modern imaging techniques, such as IVC-SCT, must be applied in difficult cases.
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Affiliation(s)
- Mari Sonoda
- Department of Pediatric Surgery, Kitano Hospital, The Tazuke Kofukai Medical Research Institute, 2-4-20 Ohgimachi, Kita-ku, Osaka, 530-8480, Japan.
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19
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Stone-containing periampullary duodenal duplication cyst with aberrant pancreatic duct. J Pediatr Surg 2009; 44:e33-5. [PMID: 19159708 DOI: 10.1016/j.jpedsurg.2008.10.106] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/29/2008] [Revised: 09/28/2008] [Accepted: 10/26/2008] [Indexed: 02/07/2023]
Abstract
This case report describes a unique variant of a duodenal duplication cyst in an 8-year-old boy who had recurrent episodes of upper abdominal pain. Imaging revealed a stone-containing cyst near the biliaropancreatic ducts and concomitant hydrops of the gallbladder. Open transduodenal marsupialization of the cyst and resection of the inflamed gallbladder were performed. Further episodes of pancreatitis did not occur for a follow-up of 2 years.
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20
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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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21
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Groebli Y, Meyer JL, Tschantz P. Choledochocele demonstrated by computed tomographic cholangiography: report of a case. Surg Today 2001; 30:272-6. [PMID: 10752782 DOI: 10.1007/s005950050058] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
Abstract
Choledochocele is a rare cystic congenital malformation of the distal common bile duct, the precise pathogenesis of which remains unclear. It is usually diagnosed in adulthood after many examinations for unclear digestive symptoms, as in the patient whose case is described herein. Of all the diagnostic techniques available, endoscopic retrograde cholangiography (ERCP) seems to be the best; however, it is an invasive procedure associated with some morbidity. Spiral computed tomographic cholangiography with three-dimensional reconstruction is also an interesting screening technique. Malignant transformation of a choledochocele occurs very rarely, compared with other cystic malformations of the biliary tract. If a choledochocele is small and symptomatic, the lesion can be treated by endoscopic sphincterotomy, but larger cysts are more effectively removed by a surgical cystoduodenostomy. We present herein the case of a 25-year-old woman admitted to our hospital in March 1998 with upper right quadrant abdominal pain caused by a choledochocele, who was successfully treated by a cystoduodenostomy.
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Affiliation(s)
- Y Groebli
- Department of Surgery, Hôpital des Cadolles, Neuchâtel, Switzerland
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Abstract
Choledochal cysts are rare developmental malformations of the biliary tree. Percutaneous and endoscopic ultrasound, as well as endoscopic retrograde cholangiopancreatography, are recommended diagnostic tools. Magnetic resonance cholangiography may also contribute to the workup and treatment plan of patients with choledochal cysts. We report a 25-yr-old white woman with episodic nausea and moderate epigastric discomfort. Magnetic resonance cholangiography showed a rather spherical, cyst-like, fluid-filled structure in continuity with the prepapillary segment of the common bile duct, thus making a choledochocele (type III choledochal cyst) likely. The patient was treated by endoscopic sphincterotomy and, after a 6-month follow-up, is without complaints.
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Affiliation(s)
- H E Adamek
- Department of Internal Medicine C, Klinikum Ludwigshafen, Academic Teaching Hospital of the University of Mainz, Germany
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23
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Huang SP, Wang HP, Chen JH, Wu MS, Shun CT, Lin JT. Clinical application of EUS and peroral cholangioscopy in a choledochocele with choledocholithiasis. Gastrointest Endosc 1999; 50:568-71. [PMID: 10502185 DOI: 10.1016/s0016-5107(99)70087-8] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/07/2023]
Affiliation(s)
- S P Huang
- Departments of Internal Medicine, Emergency Medicine, and Pathology, College of Medicine, National Taiwan University, Taipei
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Elton E, Hanson BL, Biber BP, Howell DA. Dilated common channel syndrome: endoscopic diagnosis, treatment, and relationship to choledochocele formation. Gastrointest Endosc 1998; 47:471-8. [PMID: 9647371 DOI: 10.1016/s0016-5107(98)70247-0] [Citation(s) in RCA: 21] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/08/2023]
Abstract
BACKGROUND Choledochoceles (type III biliary cysts) are cystic dilations of the terminal common bile duct or common pancreatobiliary channel. Although no size criteria have been defined, it is generally assumed these must be large. However, we describe patients who do not meet the perceived size criteria for choledochoceles, but who nonetheless have a dilated common pancreatobiliary channel. METHODS We reviewed the presenting symptoms, endoscopic and radiographic findings, and response to endoscopic therapy of patients meeting our criteria for the dilated common channel syndrome. RESULTS Of 2847 patients undergoing ERCP, 100 (3.5%) had the dilated common channel syndrome. Common presenting symptoms and signs included abdominal pain in 97%, abnormal liver function test(s) in 66%, and a history of acute or recurrent pancreatitis in 46%. A bulge was visible above the papilla in 88%, with a dilated common bile duct in 54% and a dilated pancreatic duct in 28%. After endoscopic unroofing of the common channel, 77% had complete and long-lasting resolution of symptoms, 18% had partial or transient improvement, and 5% had no change. CONCLUSIONS Although classic choledochoceles are rare, a lesser degree of dilation of the common channel is more frequent than generally appreciated. We postulate that this finding represents an "incomplete," acquired form of choledochocele, possibly caused by underlying papillary stenosis. Whatever the etiology and appropriate term, the presence of a dilated common channel predicts a high rate of clinical response to endoscopic therapy.
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Affiliation(s)
- E Elton
- Department of Medicine/Division of Gastroenterology, Maine Medical Center, Portland, USA
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