Editorial
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World J Gastroenterol. Sep 14, 2006; 12(34): 5440-5446
Published online Sep 14, 2006. doi: 10.3748/wjg.v12.i34.5440
Sporadic versus hereditary gastrinomas of the duodenum and pancreas: Distinct clinico-pathological and epidemiological features
Martin Anlauf, Nele Garbrecht, Tobias Henopp, Anja Schmitt, Regina Schlenger, Andreas Raffel, Markus Krausch, Oliver Gimm, Claus F Eisenberger, Wolfram T Knoefel, Henning Dralle, Paul Komminoth, Philipp U Heitz, Aurel Perren, Günter Klöppel
Martin Anlauf, Nele Garbrecht, Tobias Henopp, Regina Schlenger, Günter Klöppel, Department of Pathology, University of Kiel, Germany
Anja Schmitt, Paul Komminoth, Philipp U Heitz, Aurel Perren, Department of Pathology, University of Zürich, Switzerland
Regina Schlenger, Department of Forensic Medicine, University of Kiel, Germany
Andreas Raffel, Markus Krausch, Claus F Eisenberger, Wolfram T Knoefel, Department of General, Visceral and Pediatric Surgery, University of Düsseldorf, Germany
Oliver Gimm, Henning Dralle, Department of General and Visceral Surgery, University of Halle-Wittenberg, Germany
Paul Komminoth, Department of Pathology, Kantonsspital Baden, Switzerland
Supported by the Hensel Stiftung Kiel (F370011, MA and GK), the Swiss National Foundation (SNF 31-61884, AP and PK) and the German Society of Pathology (MA). Tobias Henopp has a fellowship sponsored by Ipsen GMBH, Ettlingen, Germany
Correspondence to: Dr. Martin Anlauf, Department of Pathology, University of Kiel, Michaelisstr.11, 24105 Kiel, Germany. manlauf@path.uni-kiel.de
Telephone: +49-431-5971138 Fax: +49-431-5973462
Received: June 11, 2006
Revised: June 28, 2006
Accepted: July 7, 2006
Published online: September 14, 2006
Abstract

Gastrinomas are defined as gastrin secreting tumors that are associated with Zollinger-Ellison syndrome (ZES). ZES is characterized by elevated fasting gastrin serum levels, positive secretin stimulation test and clinical symptoms such as recurrent peptic ulcer disease, gastroesophageal reflux disease and occasional diarrhea. Genetically, nonhereditary (sporadic) gastrinomas are distinguished from hereditary gastrinomas, which are associated with multiple endocrine neoplasia type 1 (MEN1) syndrome. In general, duodenal gastrinomas are small and solitary if they are sporadic and multiple as well as hereditary. The sporadic gastrinomas occur in the duodenum or in the pancreas while the hereditary gastrinomas almost all occur in the duodenum. Our series of 77 sporadic duodenal neuroendocrine tumors (NETs) includes 18 patients (23.4%) with gastrinomas and ZES. Of 535 sporadic NETs in the pancreas collected from the NET archives of the departments of pathology in Zürich, Switzerland, and Kiel, Germany, 24 patients (4.5%) suffered from sporadic pancreatic gastrinomas and ZES. These NETs have to be distinguished from tumors with immunohistochemical positivity for gastrin but without evidence of ZES. An additional 19 patients suffered from MEN1 and ZES. These patients showed exclusively duodenal gastrinomas, but not pancreatic gastrinomas. The prognosis of sporadic and MEN1-associated duodenal gastrinomas is better than that of pancreatic gastrinomas, since they progress slowly to liver metastasis. In summary, sporadic and MEN1-associated gastrinomas in the duodenum and pancreas show different clinico-pathological and genetic features. The incidence of sporadic duodenal gastrin-producing tumors is increasing, possibly due to optimized diagnostic procedures. In contrast, pancreatic MEN1-associated gastrinomas seem to be extremely rare. A considerable subset of tumors with immunohistochemical expression of gastrin but without evidence of ZES should be designated as functionally inactive NETs expressing gastrin, but not as gastrinomas.

Keywords: Endocrine tumor; Gastrinoma; Multiple endocrine neoplasia type 1; Precursor lesion; Zollinger-Ellison syndrome