Clin J Gastroenterol (2013) 6:476–479 DOI 10.1007/s12328-013-0428-4

CASE REPORT

Ductal branch-oriented pancreatic resection for an intraductal papillary mucinous neoplasm in the uncinate process that caused recurrent acute pancreatitis: a case report of successful treatment Toshiyuki Natsume • Takashi Maruyama • Akitoshi Kobayashi • Shinichiro Shimizu • Hajime Tanaka • Hiroshi Matsuzaki • Akinari Miyazaki • Takumi Ohta • Yayoi Sato • Yuji Yamamoto • Mizuho Aikawa • Ryota Otsuka • Akitoshi Yanagihara

Received: 22 May 2013 / Accepted: 9 September 2013 / Published online: 27 September 2013 Ó Springer Japan 2013

Abstract Acute pancreatitis reportedly occurs in about 15 % of cases of branch duct (BD)-intraductal papillary mucinous neoplasms (IPMNs), with two-thirds of BD-IPMNs being located in the head or uncinate process of the pancreas. However, the surgical indications and optimal treatment methods for BD-IPMNs have not been established. A 59-year-old Japanese male with epigastralgia was admitted to our hospital. A multidetector row computed tomography (MDCT) scan disclosed grade I acute pancreatitis. Magnetic resonance cholangiopancreatography disclosed a 1.5-cm BD-IPMN in the uncinate process. Two months after discharge, the epigastralgia recurred, and MDCT again revealed grade I pancreatitis. Due to the repeated episodes of pancreatitis, we performed ductal branch-oriented pancreatic resection. To detect the inferior branch of the Wirsung duct and avoid the development of a pancreatic fistula, we injected indigo carmine into the tumor which confirmed ligation of the inferior branch. Histopathologically, the tumor proved to be an adenoma. The postoperative course was uneventful in both the shortand long-term follow-up and, to date, there has been no recurrence of pancreatitis, or diabetes mellitus during the 6 T. Natsume (&)  T. Maruyama  H. Tanaka  H. Matsuzaki  A. Miyazaki  T. Ohta  Y. Sato  Y. Yamamoto  M. Aikawa  R. Otsuka  A. Yanagihara Department of Surgery, Funabashi Municipal Medical Center, 1-21-1 Kanasugi, Funabashi, Chiba 273-8588, Japan e-mail: [email protected] A. Kobayashi Department of Internal Medicine, Funabashi Municipal Medical Center, Funabashi, Japan S. Shimizu Department of Pathology, Funabashi Municipal Medical Center, Funabashi, Japan

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years since pancreatectomy. This procedure is one of the methods that can be used for the successful resection of a BD-IPMN in the uncinate process that caused recurrent acute pancreatitis. Keywords process

Acute pancreatitis  IPMN  Uncinate

Abbreviations IPMN Intraductal papillary mucinous neoplasm MDCT Multidetector row computed tomography AP Acute pancreatitis PD Pancreaticoduodenectomy

Introduction Acute pancreatitis (AP) has been reported to be caused mainly by gallstones and alcohol abuse [1]. However, there is increasing evidence that intraductal papillary mucinous neoplasms (IPMN) can also lead to pancreatitis [2, 3]. Acute pancreatitis reportedly occurs in about 15 % of cases of branch duct (BD)-IPMNs, with two-thirds of BD-IPMNs being located in the head or uncinate process of the pancreas [4]. However, the surgical indications and optimal treatment methods for BD-IPMNs have not been established. Here we report the successful resection of a case of BD-IPMN in the uncinate process that caused recurrent acute pancreatitis in a 59-year-old male who was surgically treated by ductal branch-oriented pancreatic resection. The postoperative course was uneventful in the short- and longterm follow-up and, to date, there has been no recurrence of pancreatitis, or diabetes mellitus during the 6 years since pancreatectomy. BD-IPMNs frequently have a low

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malignant potential and are usually located in the head of the pancreas [4]. We also discuss the clinical problems of treating patients with BD-IPMNs that caused recurrent acute pancreatitis, particularly those existing in the head of the pancreas.

Case report A 59-year-old Japanese male with epigastralgia was admitted to our hospital in June 2006. He had suffered an acute myocardial infarction in 2003 and had been taking medication for hyperlipidemia. He was an inveterate smoker and drank little alcohol. Laboratory studies revealed a serum amylase level of 2,652 mg/dl, urinary amylase level of 14,170 mg/dl, lipase level of 1,540 mg/dl and a white blood cell count of 13,900/ll at presentation. A multidetector row computed tomography (MDCT) scan showed fluid collection around the pancreas and diagnosed grade I acute pancreatitis. Magnetic resonance cholangiopancreatography (MRCP) revealed a 1.5-cm BD-IPMN in the uncinate process and no gallstones (Fig. 1). Endoscopic retrograde cholangiopanreatography (ERCP) revealed a 1.5-cm BD-IPMN in the uncinate process with mucin in the tumor; however, there was no mucus exuding from the major or minor papilla (Fig. 2). The patient recovered under conservative management with medication and was discharged after a 14-day hospital stay. Two months after discharge, the epigastralgia recurred, and MDCT again revealed grade I pancreatitis. He recovered under conservative management with medication and was again discharged after 14 days of hospitalization. The patient had no alcohol abuse, gallstones, or pancreas divisum and there was no other reason for the pancreatitis except the BDIPMN. Due to the repeated episodes of pancreatitis, we planned a pancreas resection. There were no abdominal

Fig. 2 ERCP revealed a 1.5-cm BD-IPMN with mucin in the tumor in the uncinate process. There was no pancreas divisum

findings in the levels of tumor markers, such as CEA, CA19-9, and DUPAN2. Endoscopic ultrasonography revealed no nodules in the BD-IPMN. We finally diagnosed the IPMN as having no malignant potential and performed ductal branch-oriented pancreatic resection in January 2007. To detect the inferior branch of the Wirsung duct and avoid the development of a pancreatic fistula, we injected indigo carmine into the tumor to avoid spillage of mucinous material from the tumor (Fig. 3a, b) and confirmed ligation of the inferior branch. The intraoperative pathologic examination revealed no invasion of the IPMN along the main pancreatic duct (Fig. 4) and no malignant potential. Histopathologically, the tumor proved to be an adenoma (Fig. 5a, b). The patient did not develop a pancreatic fistula, and his recovery followed a satisfactory postoperative course. He was dischared from our hospital in February 2007 and was carefully followed up as an outpatient. To date, there has been no recurrence of pancreatitis, or diabetes mellitus.

Discussion

Fig. 1 MRCP revealed a 1.5-cm BD IPMN in the uncinate process

As BD-IPMNs frequently have a low malignant potential, we selected reductive surgery (ductal branch-oriented pancreatic resection) for our patient who experienced repeated episodes of acute pancreatitis caused by IPMN. However, the use of reductive surgery for pancreatic tumors should be carefully considered by taking into account the safety of the procedure and the presence of negative surgical margins. Moreover, IPMNs often recur, so the validity of the reductive surgery in IPMNs is not confirmed. Although pancreaticoduodenectomy (PD) can be used in place of reductive surgery, PD results in a significant loss of the normal pancreatic parenchyma and

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Fig. 3 The tumor in the uncinate process before (a) and after (b) indigo carmine was injected

Fig. 4 The intraoperative pathologic examination revealed the stump of the pancreatic duct to be normal

increases the risk of complications like diabetes mellitus and cholangitis. Therefore, PD should be avoided if lymphadenectomy is not necessary. In our patient, we performed the reduction procedure and a close follow-up. The results revealed tumor-free margins and no complications. There has been no recurrence of the pancreatitis and IPMN during the 6 years since the procedure. Therefore, this procedure appears to be useful for BD-IPMNs located in the head of the pancreas that cause repeated episodes of acute pancreatitis. Yamaguchi et al. [5] first reported ductal branch-oriented minimal pancreatectomy. They made some recommendations to consider when performing a limited resection—clear identification of the communication duct and definite ligation of the resected line of the pancreas. In one of their patients, indigo carmine was injected directly into the cystic lesion, and successfully indicated the

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communicating points between the Wirsung duct and the inferior branch. We injected indigo carmine into the tumor according to this procedure. There are, however, several reported cases of pseudomyxoma peritonei in IPMNs [6]. Hirooka et al. [7] also reported a case of IPMN in which endosonography-guided fine needle aspiration biopsy caused dissemination. Therefore, it is essential to avoid spillage of mucinous material when injecting indigo carmine into the tumor. In general, pancreatic leakage rates up to 40 % have been reported during segmental pancreatic resection [8– 10]. Moreover, IPMNs are characterized by a growth pattern involving the spread of the tumor along the epithelium of the ductal tree [11]. Therefore, it is important to avoid damaging the Wirsung duct and to confirm no cancer cells remain at the ductal margin when a limited resection is performed. Paik et al. [12] used intestinal obstruction

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479 Disclosures Conflict of Interest: The authors declare that they have no conflict of interest. Human/Animal Rights: All procedures followed were in accordance with the ethical standards of the responsible committee on human experimentation (institutional and national) and with the Helsinki Declaration of 1975, as revised in 2008(5) Informed Consent: Informed consent was obtained from all patients for being included in the study.

References

Fig. 5 a Cut surface of the tumor. b Histopathologically, the tumor proved to be an adenoma

ultrasonography, and Takada et al. [13] used preoperative pancreatic duct stents guided by ERCP to avoid pancreatic leakage. We safely performed ductal branch-oriented pancreatic resection in our patient using indigo carmine and were thereby able to avoid a pancreatic fistula and confirm the presence of a tumor-negative margin during the operation; this therefore appears to be a useful treatment option for this type of tumor. According to the International Consensus Guidelines [14] ‘worrisome features’ on imaging include cyst of [3 cm, thickened enhanced cyst walls, and a main pancreatic duct size of C10 mm. For amelioration of symptoms, all symptomatic cysts should be further evaluated or resected as determined by the clinical circumstances. Our case had no ‘worrisome features’ but did have repeated acute pancreatitis. We therefore selected reductive surgery. In our case, we considered whether BD-IPMN caused the recurrent acute pancreatitis because there was no mucus exuding from the major or minor papilla. However, the patient had no alcohol abuse, gallstones, or pancreas divisum and there was no other reason for the pancreatitis except the BD-IPMN and the mucin in the tumor revealed by the ERCP. Therefore, we decided to perform surgery and there has been no recurrence of the pancreatitis during the 6 years since the procedure. In conclusion, we presented a case that successfully underwent ductal branch-oriented pancreatic resection of an IPMN in the uncinate process that caused recurrent acute pancreatitis. This procedure is one of the methods that can be used for the successful resection of such tumors.

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Ductal branch-oriented pancreatic resection for an intraductal papillary mucinous neoplasm in the uncinate process that caused recurrent acute pancreatitis: a case report of successful treatment.

Acute pancreatitis reportedly occurs in about 15 % of cases of branch duct (BD)-intraductal papillary mucinous neoplasms (IPMNs), with two-thirds of B...
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