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Central pancreatectomy in surgical treatment of pancreatic insulinoma--a case report /Centralna pankreatektomija uhirurskom tretmanu insulinoma pankreasa--prikaz slucaja.

UDK 616.37-006.03-089.87

Introduction

Pancreatic neuroendocrine tumors (pancreatic NETs) are uncommon neuroendocrine neoplasms that are believed to arise from pancreatic endocrine cells. Pancreatic NETs are also called islet cell tumors, because the endocrine cells of the pancreas are commonly located in clusters, anatomically called islets of Langerhans [1]. The functional pancreatic NETs are well-differentiated neoplasms with very diverse presentations related to their ability to produce and secrete hormones or peptides, such as insulin, gastrin, glucagon, vasoactive intestinal polypeptide (VIP), and somatostatin, causing characteristic hormonal syndromes [2, 3]. Of these functional tumors, up to 70% are insulinomas. Insulinomas are typically solitary tumors small in size (<1 cm). Approximately 90% of insulinomas are benign lesions that are almost always (>99%) intrapancreatic in location [4]. Insulinoma patients usually present with symptoms of hypoglycemia. Hypoglycemia is defi ned by Whipple's triad or Whipple's criteria consi sting of central nervous system symptoms of neuro glycopenia, a simultaneous low blood glucose level and relief of the symptoms after administration o glucose [5]. Central pancreatectomy was first descri bed and performed by Guillemin and Bessot in 1957 in the treatment of chronic pancreatitis. This surgica procedure is currently reserved for selective mana gement of benign lesions located in the pancreatic neck and body. The main benefit of central pancre atectomy is the preservation of pancreatic endocrine and exocrine function, as well as spleen preservation that is important especially in younger patients [6].

Case report

We present a case of sporadic insulinoma in a yo ung, 24-year-old female patient, who presented witl a 2-month history of episodic shaking, diaphoresis increased hunger, confusion, obtundation and fainting Symptoms of neuroglycopenia were predominant, sc the family members reported that the patient has un dergone a personality change. Subsequently, the patient was admitted to the Psychiatry Clinic, Clinica Center of Vojvodina. Initial laboratory investigation: showed low blood glucose of 1.8 mmol/L (normal ran ge, 4.4 to 6.1 mmol/L). Due to repeatedly low blood glucose levels, an endocrinologist was consulted. The patient was then admitted to the Clinic of Endocrino logy for further examination as a possible case of pancreatic insulinoma. A 72-hour mentored fasting test was done, during which blood was drawn and results showed low blood glucose level, (2.1 mmol/L), elevated insulin of 74.6 mU/l (normal range, 3-25 mU/l) and C-peptide level of 8.4 ng/ml (normal range, 0.8-3.9 ng/ ml). The hypoglycemic symptoms were relieved after glucose administration. The patient underwent an abdominal contrast-enhanced computed tomography (CECT) scan that showed a 12 x 15 x 17 mm, well-demarcated, heterogeneous lesion within the body of the pancreas (Figure 1). Locoregional lymphadenopathy and liver metastasis were excluded by CECT. The patient was then admitted to the Clinic of Abdominal, Endocrine and Transplantation Surgery, Clinical Center of Vojvodina, and underwent surgical treatment. Central pancreatectomy was performed, with complete removal of the tumor (Figure 2), and conservation of head and distal pancreas. The main pancreatic duct within the head of the pancreas was individually ligated with a permanent suture, and posterior pancreatic capsules were approximated with full-thickness interrupted absorbable sutures (Figure 3). Anterior and posterior wall of pancreas were approximated with full-thickness interrupted absorbable sutures. The reconstruction of pancreaticoenteric anastomosis was accomplished with a retrocolic, Rouxen-Y pancreaticojejunostomy (Figure 4). The specimen of resected pancreatic tissue was transected to show a well-demarcated lesion (Figure 5). Histological evaluation and immunohistochemical examination of the specimen confirmed a pancreatic NET--insulinoma. The patient had an uneventful recovery. The post-operative follow-up showed resolution of hypoglycemic symptoms and normal blood glucose and insulin levels. Computed tomography (CT) imaging showed no evidence of tumor recurrence.

Discussion

Insulinomas are functional pancreatic NETs which secrete endogenous insulin autonomously, regardless of blood glucose levels, resulting in a state of hyperinsulinemia [7]. In the majority of cases they present as solitary sporadic tumors localized in the pancreas, but they can also be part of multiple endocrine neoplasia type 1 (MEN 1) [4]. Insulinomas are generally small tumors (<1 cm) that are best located by using endoscopic ultrasound (EUS). Although the majority of insulinomas have a benign course and can be managed by surgery, it is important to diagnose large tumors with possible liver metastases using CT or magnetic resonance imaging (MRI), because it may completely change the surgical strategy [3].

Pancreatic leak is the most common complication of this surgical technique. The pancreaticoenteric anastomosis is the weak point of every pancreatic resection that includes drainage of the pancreatic remnant to the gastrointestinal tract. The majority of pancreatic leaks have a benign course and do not require any intervention, except long-term maintenance of intra-operatively placed drains [8]. In complicated cases, pancreatic leaks may lead to formation of abscess and/ or destruction of the surrounding tissues. If erosion of blood vessels occurs, it can cause severe hemorrhage, which is the major cause of postoperative mortality [9]. In our case, the exocrine function of the pancreas was reestablished with a retrocolic, Roux-en-Y pancreaticojejunostomy. For more common pancreatoduodenectomies, we routinely perform a 2-layer, end-toside, duct-to-mucosa pancreaticojejunostomy with a free-floating stent [10]. Preserved exocrine and endocrine function of pancreas are major benefits of this surgical technique. Meta-analysis by Iacono et al., which included two prospective and nineteen retrospective studies emphasize the significance of preserved exocrine and endocrine function in central pancreatectomy, in comparison to distal pancreatectomy [11]. Although technically demanding and not always feasible, spleen preservation is a major immunological advantage. Patients without a spleen have an impaired phagocytosis and opsonization, as well as decreased levels of immunoglobulins. Overwhelming postsple nectomy infection (OPSI) syndrome is a rare but serious complication of splenectomy, with mortality rates approaching 50% [12].

Conclusion

Insulinomas are rare pancreatic neuroendocrine tumors, but they should always be considered in the differential diagnosis of any patient presenting with frequent hypoglycemic symptoms. Surgical removal through enucleation or segmental resection of the pancreas is the mainstay of therapy. Central pancreatectomy may preserve endocrine and exocrine function, as well as the immunological role of the spleen. It is a safe surgical technique with low mortality, but potentially high complication rate. The most common complication is pancreatic leak. Central pancreatectomy is a demanding surgical procedure and should only be performed at high-volume centers by experienced surgeons.

Abbreviations
Pancreatic NETs   --pancreatic neuroendocrine tumors
CECT              --contrast-enhanced computed tomography
CT                --computed tomography


DOI: 10.2298/MPNS1704111M

References

[1.] Ong SL, Garcea G, Pollard CA, Furness PN, Steward WP, Rajesh A, et al. A fuller understanding of pancreatic neuroendocrine tumours combined with aggressive management improves outcome. Pancreatology. 2009;9(5):583-600.

[2.] Okabayashi T, Shima Y, Sumiyoshi T, Kozuki A, Ito S, Ogawa Y, et al. Diagnosis and management of insulinoma. World J Gastroenterol. 2013;19(6):829-37.

[3.] National Comprehensive Cancer Network. NCCN clinical practice guidelines in oncology version 1.2012: neuroendocrine tumors. [cited 2012 May 10]. Available from: http://www.nccn.org/ professionals/physician_gls/pdf/neuroendocrine.pdf

[4.] Kulke MH, Anthony LB, Bushnell DL, de Herder WW, Goldsmith SJ, Klimstra DS, et al. NANETS treatment guidelines: well-differentiated neuroendocrine tumors of the stomach and pancreas. Pancreas. 2010;39(6):735-52.

[5.] Cryer PE, Axelrod L, Grossman AB, Heller SR, Montori VM, Seaquist ER, et al. Evaluation and management of adult hypoglycemic disorders: an Endocrine Society Clinical Practice Guideline. J Clin Endocrinol Metab. 2009;94(3):709-28.

[6.] Christein JD, Smoot RL, Farnell MB. Central pancreatectomy: a technique for the resection of pancreatic neck lesions. Arch Surg. 2006;141(3):293-9. Rad je primljen 24. I 2017. Recenziran 16. II 2017. Prihvacen za stampu 18. II 2017. BIBLID.0025-8105:(2017):LXX:3-4:111-114.

[7.] Abbasakoor NO, Healy ML, O'Shea D, Maguire D, Muldoon C, Sheahan K, et al. Metastatic insulinoma in a patient with type 2 diabetes mellitus: case report and review of the literature. Int J Endocrinol. 2011;2011:124078.

[8.] Ho CK, Kleeff J, Friess H, Buchler MW. Complications of pancreatic surgery. HPB (Oxford). 2005;7(2):99-108.

[9.] Berberat PO, Friess H, Kleeff J, Uhl W, Buchler MW. Prevention and treatment of complications in pancreatic cancer surgery. Dig Surg. 1999;16(4):327-36.

[10.] Milosevic P, Bolaji M, Milosevic B, Ikonic N, Popovic A, Veljkovic R. Uloga hirurske resekcije u tretmanu adenokarcinoma pankreasa. Med Pregl. 2011;64(9-10):448-52.

[11.] Iacono C, Verlato G, Ruzzenente A, Campagnaro T, Bacchelli C, Valdegamberi A, et al. Systematic review of central pancreatectomy and meta-analysis of central versus distal pancreatectomy. Br J Surg. 2013;100(7):873-85.

[12.] Bisharat N, Omari H, Lavi I, Raz R. Risk of infection and death among post-splenectomy patients. J Infect. 2001;43 (3):182-6.

Uros MILOSEVIC (1,2), Aleksandar GLUHOVIC (1,2), Dorde MILOSEVIC (1,2), Nebojsa BUDAKOV (1,3) and Pavle MILOSEVIC (1,2)

University of Novi Sad, Faculty of Medicine, Department of Surgery (1) Clinical Center of Vojvodina, Novi Sad Clinic of Abdominal, Endocrine and Transplantation Surgery (2) Clinic of Vascular and Transplantation Surgery (3)

Corresponding Author: Dr Uros Milosevic, Klinicki centar Vojvodine, Klinika za abdominalnu, endokrinu i transplantacionu hirurgiju, 21000 Novi Sad, Hajduk Veljkova 1-7, E-mail: urospmilosevic@gmail.com

Caption: Figure 1. Axial view of the abdominal CECT scan: showing 12 x 15 x 17 mm well demarcated lesion located within the neck of the pancreas (arrow)

Slika 1. Aksijalni presek kontrastom poboljsane kompjuterizovane tomografije: prikazuje 12 x 15 x 17 mmjasno ogranicenu promenu lokalizovanu u vratu pankreasa (strelica)

Caption: Figure 2. Intraoperative view showing the insulinoma in the pancreatic body (white arrow)

Slika 2. Intraoperativni prikaz insulinoma tela pankreasa (bela strelica)

Caption: Figure 3. Intraoperative view showing the preserved head of the pancreas after central pancreatectomy (white arrow) Slika 3. Intraoperativni prikaz prezervirane glave pankreasa nakon centralne pankreatektomije (bela strelica)

Caption: Figure 4. Intraoperative view after Roux-en-Y, end-toend pancreaticojejunostomy (white arrow)

Slika 4. Intraoperativni prikaz nakon Roux-en-Y, terminoterminalne pankreatikojejunostomije (bela strelica)

Caption: Figure 5. The transected specimen of the resected pancreatic tissue with well-demarcated lesion--insulinoma

Slika 5. Transecirani preparat reseciranog tkiva pankreasa sa jasno ogranicenom promenom--insulinom
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Title Annotation:CASE REPORTS PRIKAZISLUCAJEVA
Author:Milosevic, Uros; Gluhovic, Aleksandar; Milosevic, Dorde; Budakov, Nebojsa; Milosevic, Pavle
Publication:Medicinski Pregled
Article Type:Case study
Date:Mar 1, 2017
Words:1654
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