Case Reports in Medicine

Case Reports in Medicine / 2018 / Article

Case Report | Open Access

Volume 2018 |Article ID 3258141 |

Ryuji Komine, Atsushi Shimizu, Kazuhiko Mori, Keisuke Minamimura, Toru Hirata, Takashi Kobayashi, Nobuo Toda, Masaya Mori, "Pancreaticoduodenectomy for an Ampullary Region Carcinoma Occurred in Annular Pancreas Coexistent with Replaced Common Hepatic Artery", Case Reports in Medicine, vol. 2018, Article ID 3258141, 5 pages, 2018.

Pancreaticoduodenectomy for an Ampullary Region Carcinoma Occurred in Annular Pancreas Coexistent with Replaced Common Hepatic Artery

Academic Editor: William B. Silverman
Received14 Dec 2017
Accepted23 Jan 2018
Published20 Feb 2018


Introduction. Annular pancreas is a rare congenital abnormality characterized by a ring of pancreatic tissue surrounding the descending portion of the duodenum. Annular pancreas coexisting with replaced common hepatic artery which is also a rare anatomical variation has not been reported previously. Case Presentation. A 53-year-old man visited our hospital complaining of epigastric pain. Based on radiological examinations, he was diagnosed as having pancreatitis, annular pancreas, and hepatomesenteric trunk. One month later, obstructive jaundice developed. Endoscopic examination revealed ampullary region carcinoma. We performed pancreaticoduodenectomy using the “artery-first” approach. Discussion. Both annular pancreas and common hepatic artery anomaly are rare. High-quality preoperative imaging and awareness of such rare conditions are necessary for operative safety. Although the embryological relationship between these anomalies is uncertain, the present case may suggest some relevance between the two. Conclusion. The “artery-first” approach may be a useful method for pancreaticoduodenectomy in patients who have an anatomical abnormality.

1. Introduction

Annular pancreas (AP) is a rare congenital anomaly in which the second part of the duodenum is surrounded by a ring of pancreatic tissue continuous with the head of the pancreas. The prevalence of AP was reported in 3 of 20,000 autopsies and 3 of 24,519 surgical cases [1, 2]. Although pancreatitis, duodenal obstruction, and so on are known as symptoms of AP, approximately two-thirds of the patients are asymptomatic [3]. AP is also regarded as one of the risk factors of pancreatobiliary malignancy. However, replaced common hepatic artery (CHA) arising from the superior mesenteric artery (SMA) was reportedly seen in 1.13% of 19,013 cases [4]. The hepatomesenteric trunk is also rarely encountered but can be problematic in pancreaticoduodenectomy (PD). Although the relationship between these rare anomalies is uncertain, embryologically, they are both considered to occur during the same week of gestation [5]. We herein describe an extremely rare case in which these anomalies coexisted in a patient with ampullary region carcinoma treated by PD with the “artery-first” approach.

2. Case Presentation

A 53-year-old man presented with epigastric pain visited our hospital. Result of the blood analysis indicated hyperamylasemia. The abdominal computed tomography (CT) scan showed diffuse parenchymal enlargement, and he was diagnosed as having mild pancreatitis according to the modified CT severity index. The CT scan also revealed an anomaly of the CHA arising from the SMA (Figure 1(a)) and annular pancreatic duct. Magnetic resonance cholangiopancreatography (MRCP) confirmed that the annular duct formed a loop in the head of the pancreas (Figure 1(b)). There were no pancreaticobiliary maljunction. Neither the common bile duct nor the main pancreatic duct was dilated. Endoscopic ultrasonography was performed to investigate other causes, but the ampulla of Vater could not be observed due to technical difficulty.

One month later, result of the follow-up laboratory test demonstrated obstructive jaundice. The CT scan revealed a dilatation of the common bile duct. An ulcerative tumor was found around the duodenal papilla region by upper esophagogastroduodenoscopy (Figure 1(c)). Biopsy results from the ulcer indicated adenocarcinoma. Endoscopic retrograde cholangiography (ERCP) was performed to investigate the biliary or pancreatic duct, but cannulation of the ampulla of Vater could not be performed successfully due to the invasion of the tumor.

Subtotal stomach-preserving pancreaticoduodenectomy with dissection of the regional lymph nodes was performed. Laparotomy showed that the pancreatic parenchymal tissue surrounded the descending part of the duodenum. Neither liver metastasis nor peritoneal dissemination was found. The tumor was located in the head of the pancreas. The SMA was exposed first to identify the origin of the CHA (the “artery-first” approach). The CHA arouse from posteriorly to pancreatic head and traveled posteriorly to the portal vein (PV), and then it ran anteriorly to the PV after it branched off the gastroduodenal artery and anterior superior pancreaticoduodenal artery (Figure 2). The pancreas was transected at the neck directly overlying the PV-SMV-splenic vein confluence.

Cholangiopancreatography of the resected specimen showed that the annular duct was connected to the major papilla (Figures 3(a) and 3(b)). The final pathology revealed poorly differentiated adenocarcinoma (Figure 4) without regional lymph node metastasis (International Union Against Cancer TNM classification, 8th edition: T3N0M0, IIA). The patient’s postoperative course was uneventful, and he was discharged 21 days postoperatively. He is doing well without any evidence of recurrence at the most recent follow-up of 6 months postoperatively.

3. Discussion

Recently, with modern diagnostic imaging devices, such as CT, MRCP, or ERCP, 1 in 250 examined cases was found to have AP [6, 7]. About 700 cases of AP have been reported in the literatures [8], and a relationship with the increasing incidence of pancreatobiliary malignancy also has been described [811]. Among the cases reported in the English literatures, there are eight resected cases of ampullary region carcinoma associated with AP (Table 1) [1117]. It should be important to raise the suspicion of potential malignancy when symptoms occur in adult patients who have AP and have been asymptomatic.

CaseAuthorAgeSexSymptomsOperationTNM classificationSize (cm)

1Transveldt et al. [12]80FJaundice, WLPDNDND
2Benger and Thompson [13]66MJaundice, WLPDT2N0M0ND
3Rathnaraj et al. [11]55MJaundicePDND2 × 2
4Shan et al. [14]40FJaundice, APPDNDND
5Shan et al. [14]45MJaundice, APPPPDNDND
6Foo et al. [15]78FJaundice, APPDT1N0M01.2 × 3.5
7Yazawa et al. [16]59MEDPPPDTisN0M02.5 × 2.0
8Tewari et al. [17]42FJaundice, APPDND2.0 × 2.0
9Present case53MEpigastric painSSPPDT2N0M03.3 × 3.0

PPPD: pylorus-preserving pancreaticoduodenectomy; SSPPD: subtotal stomach-preserving pancreaticoduodenectomy; WL: weight loss; AP: abdominal pain; ED: epigastric discomfort; ND: not described.

Yogi et al. classified the annular pancreas as six variants of the ductal anatomy. In accordance with this classification, type 2, in which the duct of Wirsung encircles the duodenum but drains at the major papilla, corresponds with our case [18]. ERCP is usually useful to diagnose pancreatic duct; however, in our case, the transpapillary approach or cannulation to the pancreatic duct could not be performed because of direct invasion of the carcinoma. MRCP may be another diagnostic option for AP, but it cannot always identify the minor papilla or thin Santorini duct. A contrast study of the resected specimen was the most informative. In our case, AP did not become a technical problem during the operation because the pancreas was transected at the distal side of the AP. It is also important to clearly identify the dilated main pancreas pancreatic duct to understand the path of the annular duct before the operation for safe resection.

Michels described the hepatic arterial anatomy and variations of 200 cadaveric dissections and identified 10 types of hepatic arterial anatomy [19]. Our case was classified as type IX. Accidental ligation of the hepatic arteries can lead to hepatic dysfunction, ischemia of bilioenteric anastomosis, and may result in a fatal leak. From a technical perspective, the “artery-first” approach was also useful in the case of anomaly. Sanjay et al. described types and advantages of six “artery-first” approaches [20]. We used the posterior approach; that is, we exposed and taped the root of the SMA initially in front of the left renal vein after kocherization. This enabled total control of the arterial anomaly before identification and ligation, which contributed to safe resection and secure lymph node dissection even in an extremely rare condition.

Our case may suggest some embryological relevancies between AP and hepatic arterial anomalies. The developmental mechanism of AP has not been determined, but Lecco’s theory, in which the adherence of the ventral bud to the duodenal wall prior to rotation is regarded as the causes, seems to be more accepted than other theories [21]. The hepatomesenteric trunk is considered to build due to degeneration and adhesion of ventral splanchnic arteries [22]. Both anatomical anomalies are formed during the same fifth to seventh week of gestation. Fukai et al. reported that all of the pancreatic tissue of the AP may be derived from the ventral anlage, and presented a hypothesis about Yogi’s type 2 AP based on Lecco’s theory. According to the theory, incomplete rotation, overgrowth of the tip of the ventral anlage, and adhesion to the dorsal anlage could make the annular duct fused with the main duct of the dorsal anlage instead of crossing over the lower bile duct [23]. In addition, the CHA anomaly was reported to sometimes pass through the pancreatic head and running in front of the main pancreatic duct in those cases [19, 24, 25]. In our case, the CHA passed behind the head of the pancreas, on the left side of the annular duct. It may be rational to consider that the rotation and adhesion of the pancreatic bud occur somewhat later than the formation of the vascular structure, and the vasculature is one of the regulators that determine the position of the fusion of the main pancreatic duct and annular duct.


AP:Annular pancreas
CHA:Common hepatic artery
SMA:Superior mesenteric artery
CT:Computed tomography
MRCP:Magnetic resonance cholangiopancreatography
ERCP:Endoscopic retrograde cholangiography.

Written informed consent was obtained from the patient for publication of this case report and any accompanying images.

Conflicts of Interest

The authors declare that they have no conflicts of interest.

Authors’ Contributions

Ryuji Komine and Atsushi Shimizu performed the operation, conceived and designed the study, and drafted the manuscript. Keisuke Minamimura, Kazuhiko Mori, Toru Hirata, and Takashi Kobayashi had taken charge of management of the patients. Nobuo Toda Masaya Mori had contributed significantly to the assessments and supervised other authors. All authors read and approved the final manuscript.


  1. M. M. Ravitch and A. C. Woods Jr., “Annular pancreas,” Annals of Surgery, vol. 132, no. 6, pp. 1116–1127, 1950. View at: Publisher Site | Google Scholar
  2. T. Theodorides, “Annular pancreas,” Journal of Chiropractic Education, vol. 87, pp. 445–462, 1964. View at: Google Scholar
  3. J. E. Cunha, M. S de Lima, J. Jukemura et al., “Unusual clinical presentation of annular pancreas in the adult,” Pancreatology, vol. 5, no. 1, pp. 81–85, 2005. View at: Publisher Site | Google Scholar
  4. E. Panagouli, D. Venieratos, E. Lolis, and P. Skandalakis, “Variations in the anatomy of the celiac trunk: a systematic review and clinical implications,” Annals of Anatomy-Anatomischer Anzeiger, vol. 195, no. 6, pp. 501–511, 2013. View at: Publisher Site | Google Scholar
  5. T. W. Sadler, Medical Embryology, Lippincott Williams & Wilkins, Philadelphia, PA, USA, 12th edition, 2012.
  6. B. B. Baggott and W. B. Long, “Annular pancreas as a cause of extrahepatic biliary obstruction,” American Journal of Gastroenterology, vol. 86, no. 2, pp. 224–226, 1991. View at: Google Scholar
  7. G. M. Glazer and A. R. Margulis, “Annular pancreas: etiology and diagnosis using endoscopic retrograde cholangiopancreatography,” Radiology, vol. 133, no. 2, pp. 303–306, 1979. View at: Publisher Site | Google Scholar
  8. N. J. Zyromski, J. A. Sandoval, H. A. Pitt et al., “Annular pancreas: dramatic differences between children and adults,” Journal of the American College of Surgeons, vol. 206, no. 5, pp. 1019–1025, 2008. View at: Publisher Site | Google Scholar
  9. Y. Yogi, S. Kosai, S. Higashi, T. Iwamura, and T. Setoguchi, “Annular pancreas associated with pancreatolithiasis: a case report,” Hepatogastroenterology, vol. 46, no. 25, pp. 527–531, 1999. View at: Google Scholar
  10. K. Sandrasegaran, A. Patel, E. L. Fogel, N. J. Zyromski, and H. A. Pitt, “Annular pancreas in adults,” American Journal of Roentgenology, vol. 193, no. 2, pp. 455–60, 2009. View at: Publisher Site | Google Scholar
  11. S. Rathnaraj, S. K. Singh, and M. Varghese, “Annular pancreas associated with carcinoma of papilla,” Indian Journal of Gastroenterology, vol. 17, p. 110, 1998. View at: Google Scholar
  12. E. Transveldt, R. G. Keith, J. Fonger, and M. M. Fischer, “Annular pancreas associated with coexistent ampullary carcinoma in an elderly woman,” Canadian Journal of Surgery, vol. 25, no. 6, pp. 687-688, 1982. View at: Google Scholar
  13. J. R. Benger and M. H. Thompson, “Annular pancreas and obstructive jaundice,” American Journal of Gastroenterology, vol. 92, no. 4, pp. 713-714, 1997. View at: Google Scholar
  14. Y. S. Shan, E. D. Sy, and P. W. Lin, “Annular pancreas with obstructive jaundice: beware of underlying neoplasm,” Pancreas, vol. 25, no. 3, pp. 314–316, 2002. View at: Publisher Site | Google Scholar
  15. F. J. Foo, U. Gill, C. S. Verbeke, J. A. Guthrie, and K. V. Menon, “Ampullary carcinoma associated with an annular pancreas,” Journal of the Pancreas, vol. 8, no. 1, pp. 50–54, 2007. View at: Google Scholar
  16. N. Yazawa, T. Imaizumi, D. Furukawa et al., “An annular pancreas associated with carcinoma of the papilla of Vater: report of a case,” Surgery Today, vol. 42, no. 5, pp. 497–501, 2012. View at: Publisher Site | Google Scholar
  17. M. Tewari, A. Verma, and H. Shukla, “Carcinoma of the ampulla of Vater associated with annular pancreas,” Indian Journal of Surgery, vol. 78, no. 5, pp. 409-410, 2016. View at: Publisher Site | Google Scholar
  18. Y. Yogi, T. Shibue, and S. Hashimoto, “Annular pancreas detected in adults, diagnosed by endoscopic retrograde cholangiopancreatography: report of four cases,” Gastroenterologia Japonica, vol. 22, no. 1, pp. 92–99, 1987. View at: Google Scholar
  19. N. A. Michels, “Newer anatomy of the liver and its variant blood supply and collateral circulation,” American Journal of Surgery, vol. 112, no. 3, pp. 337–347, 1966. View at: Publisher Site | Google Scholar
  20. P. Sanjay, K. Takaori, S. Govil, S. V. Shrikhande, and J. A. Windsor, “‘Artery-first’ approaches to pancreatoduodenectomy,” British Journal of Surgery, vol. 99, no. 8, pp. 1027–1035, 2012. View at: Publisher Site | Google Scholar
  21. T. M. Lecco, “Zur morphologic des pankreas annulare,” Sitzungsb Akad Wissensch, vol. 4, pp. 299–304, 1910. View at: Google Scholar
  22. Y. Wang, C. Cheng, L. Wang, R. Li, J. H. Chen, and S. G. Gong, “Anatomical variations in the origins of the celiac axis and the superior mesenteric artery: MDCT angiographic findings and their probable embryological mechanisms,” European Radiology, vol. 24, no. 8, pp. 1777–1784, 2014. View at: Publisher Site | Google Scholar
  23. M. Fukai, T. Kamisawa, S. I. Horiguchi et al., “A case of annular pancreas with Wirsung’s duct encircling the duodenum: embryological hypothesis based on cholangiopancreatographic and immunohistochemical findings,” Clinical Journal of Gastroenterology, vol. 10, no. 3, pp. 283–288, 2017. View at: Publisher Site | Google Scholar
  24. S. Yamamoto, K. Kubota, K. Rokkaku, T. Nemoto, and A. Sakuma, “Disposal of replaced common hepatic artery coursing within the pancreas during pancreatoduodenectomy: report of a case,” Surgery Today, vol. 35, no. 11, pp. 984–987, 2005. View at: Publisher Site | Google Scholar
  25. M. Mizumoto, S. Nishimura, G. Honjo, and S. Matsusue, “Pancreaticoduodenectomy for carcinoma of the ampulla of Vater with common hepatic artery originated from hepatomesenteric trunk, passing through the pancreatic head: report of a case (in Japanese),” Japanese Journal of Gastroenterological Surgery, vol. 42, no. 11, pp. 1670–1675, 2009. View at: Publisher Site | Google Scholar

Copyright © 2018 Ryuji Komine et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

More related articles

 PDF Download Citation Citation
 Download other formatsMore
 Order printed copiesOrder

Related articles

Article of the Year Award: Outstanding research contributions of 2020, as selected by our Chief Editors. Read the winning articles.