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The sun beside the pancreatic neck: The lymph node gastrinoma (with video)


1 Department of Rheumatology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China
2 Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China
3 Department of Nuclear Medicine, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China
4 Department of Pathology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China

Date of Submission24-Oct-2021
Date of Acceptance27-Feb-2022
Date of Web Publication08-Jun-2022

Correspondence Address:
Aiming Yang,
Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing
China
Shengyu Zhang,
Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing
China
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/EUS-D-21-00227



How to cite this URL:
Zhang G, Jiang Q, Zhang S, Wu D, Luo Y, Zhou W, Yang A. The sun beside the pancreatic neck: The lymph node gastrinoma (with video). Endosc Ultrasound [Epub ahead of print] [cited 2022 Jun 27]. Available from: http://www.eusjournal.com/preprintarticle.asp?id=346864

Dr. Guizhi Zhang and Qingwei Jiang contributed equally to this work and should be considered as co.first authors.


A 49-year-old male was admitted because of watery diarrhea, acid reflux, and intermittent abdominal pain. The symptoms could be alleviated by regular uptake of proton-pump inhibitors. The endoscopic examination showed the thick folds of the gastric body. The gastrin level was elevated (420 pg/mL; normal range, 25–100 pg/mL). The computed tomography (CT) scan of the abdomen revealed a 1.7-cm soft-tissue mass with enhancement adjacent to the lesser curvature of the stomach and anterior to the pancreatic neck, which also showed a high uptake in the somatostatin receptor tomography (SRT) [Figure 1]. EUS could locate this lesion (contrast-enhanced EUS, Video 1 [Additional file 1]), and fine-needle aspiration (FNA) was accomplished. The pathological finding of FNA was consistent with neuroendocrine tumor [Figure 2], while no submucosal lesion was seen in duodenum in both EUS scanning and esophagogastroduodenoscopy examination. The patient underwent laparoscopic lesser omentum mass resection. The pathological findings showed neuroendocrine tumor in lymph node (LN) [Figure 2], in which immunohistochemical staining showed chromogranin A (CgA) (+), synaptophysin (+), and Ki-67 (index 1%) [Figure 2]. The gastrin level rapidly decreased to normal (29 pg/mL) in 3 days postoperatively and remained normal in 1-month and 1-year follow-up. There was no recurrence of diarrhea after operation and PPI discontinuation. No new lesion was detected by CT and SRT in 1-year follow-up.
Figure 1: Computed tomography and somatostatin receptor tomography. (a,b) abdominal contrast computed tomography demonstrating a 1.7-cm lesion (arrow) adjacent to the lesser curvature of the stomach (anterior to the pancreatic neck); (c) the high octreotide uptake of the same lesion in somatostatin receptor tomography, looking like the “sun” beside the pancreatic neck

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Figure 2: Pathology findings of lymph node gastrinoma. (a) The hematoxylin and eosin staining of EUS-FNA sample smear shows the small round cells, whose morphology is consistent with neuroendocrine tumor; (b) gross specimen of resected lymph node gastrinoma; (c and d) immunohistochemistry staining shows positivity of chromogranin A and synaptophysin; (e and f) shows the neuroendocrine tumor cells in the lymph node

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Gastrinoma accounts for 70% of cases of pancreatic neuroendocrine tumors.[1] The majority of gastrinomas are sporadic, which usually occur in the “gastrinoma triangle” (which is composed of the porta hepatis, duodenal sweep, and pancreatic head);[2] ectopic gastrinomas are rare (<5%), which may grow in LN, liver, bile duct, and ovary.[3] Gastrin overproduction leads to oversecretion of stomach acid, causes multiple peptic ulceration, and deactivates pancreatic enzymes which result in fat malabsorption and diarrhea.[4],[5] The fasting gastrin is the preferred biochemical diagnosis test, and an increase in gastrin levels >120 pg/mL over basal fasting levels is considered positive.[6] CT and magnetic resonance imaging (MRI) are thought to have high specificity (about 95%–100%); but the sensitivity of CT for smaller tumors or tumors outside the pancreas and liver is reduced, while MRI has an advantage in detecting liver metastases. SRT improved the higher sensitivity of tumor localization to 78%–88% than 25%–85% of CT and MRI.[7] EUS is more sensitive modality for detecting small intrapancreatic tumors, commonly used for screening multiple endocrine neoplasm type 1.[8]

There is sufficient evidence that primary LN gastrinoma exists.[9] A study of 176 gastrinoma patients followed for 10 years revealed that primary LN gastrinoma was present in 10% of patients, which is not uncommon.[10] Pathological evidence for the presence of neuroendocrine cells in trigonal LNs in patients without gastrinoma supports the possibility of primary gastrinoma in LNs.[11] Tissue development studies suggest that pancreatic stem cells from the ventral buds disperse and merge into lymphoid tissue and duodenal wall during embryonic development.[12] However, there was no difference in clinical characteristics, number, and location of LNs between patients with primary and metastatic LNs gastrinoma.[13] The recommended diagnostic criteria may be used to distinguish between primary LN gastrinoma and LN metastases from unknown lesions of gastrinoma: Careful exploration without tumor in other sites during surgery, rapid normalization of the serum gastrin level after surgery, continuous normalization of serum gastrin, and the absence of disease during the postoperative follow-up period.[14] A long-term disease-free survival of more than 10 years with negative gastrin level as well as negative multiple imaging studies (CT, MRI, and SRT) argues for the likelihood that the LN gastrinoma was the primary.[10]

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published, and due efforts will be made to conceal his identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Beijing Science and Technology Program (Z181100001618013); Peking Union Medical College Education Reform Program (2019zlgc1006).

Conflicts of interest

Aiming Yang is an Editorial Board Member of the journal. The article was subject to the journal's standard procedures, with peer review handled independently of this member and his research group. There are no other conflicts of interest.

 
  References Top

1.
Jensen RT, Niederle B, Mitry E, et al. Gastrinoma (duodenal and pancreatic). Neuroendocrinology 2006;84:173-82.  Back to cited text no. 1
    
2.
Norton JA, Fraker DL, Alexander HR, et al. Surgery increases survival in patients with gastrinoma. Ann Surg 2006;244:410-9.  Back to cited text no. 2
    
3.
Yeung MJ, Pasieka JL. Gastrinomas: A historical perspective. J Surg Oncol 2009;100:425-33.  Back to cited text no. 3
    
4.
Roy PK, Venzon DJ, Shojamanesh H, et al. Zollinger-Ellison syndrome. Clinical presentation in 261 patients. Medicine (Baltimore) 2000;79:379-411.  Back to cited text no. 4
    
5.
Hirschowitz BI, Simmons J, Mohnen J. Clinical outcome using lansoprazole in acid hypersecretors with and without Zollinger-Ellison syndrome: A 13-year prospective study. Clin Gastroenterol Hepatol 2005;3:39-48.  Back to cited text no. 5
    
6.
Berna MJ, Hoffmann KM, Long SH, et al. Serum gastrin in Zollinger-Ellison syndrome: II. Prospective study of gastrin provocative testing in 293 patients from the National Institutes of Health and comparison with 537 cases from the literature. Evaluation of diagnostic criteria, proposal of new criteria, and correlations with clinical and tumoral features. Medicine (Baltimore) 2006;85:331-64.  Back to cited text no. 6
    
7.
Mendelson AH, Donowitz M. Catching the zebra: Clinical pearls and pitfalls for the successful diagnosis of Zollinger-Ellison syndrome. Dig Dis Sci 2017;62:2258-65.  Back to cited text no. 7
    
8.
van Asselt SJ, Brouwers AH, van Dullemen HM, et al. EUS is superior for detection of pancreatic lesions compared with standard imaging in patients with multiple endocrine neoplasia type 1. Gastrointest Endosc 2015;81:159-67.e2.  Back to cited text no. 8
    
9.
Cavalcanti E, Stasi E, Coletta S, et al. Primary lymph node gastrinoma: A case report and review of the literature. World J Surg Oncol 2020;18:80.  Back to cited text no. 9
    
10.
Norton JA, Alexander HR, Fraker DL, et al. Possible primary lymph node gastrinoma: Occurrence, natural history, and predictive factors: A prospective study. Ann Surg 2003;237:650-7.  Back to cited text no. 10
    
11.
Herrmann ME, Ciesla MC, Chejfec G, et al. Primary nodal gastrinomas. Arch Pathol Lab Med 2000;124:832-5.  Back to cited text no. 11
    
12.
Passaro E Jr., Howard TJ, Sawicki MP, et al. The origin of sporadic gastrinomas within the gastrinoma triangle: A theory. Arch Surg 1998;133:13-6.  Back to cited text no. 12
    
13.
Chen Y, Deshpande V, Ferrone C, et al. Primary lymph node gastrinoma: A single institution experience. Surgery 2017;162:1088-94.  Back to cited text no. 13
    
14.
Abu Ghanimeh M, Abuamr K, Sadeddin E, et al. Severe chronic diarrhoea secondary to primary lymph node gastrinoma. BMJ Case Rep 2017;2017:bcr2016216855.  Back to cited text no. 14
    


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