PMID- 29181825 OWN - NLM STAT- PubMed-not-MEDLINE LR - 20201001 IS - 2198-7793 (Print) IS - 2198-7793 (Electronic) IS - 2198-7793 (Linking) VI - 3 IP - 1 DP - 2017 Nov 28 TI - A primary hepatic gastrinoma accompanied by hyperplasia of multi-nodular Brunner's glands. PG - 118 LID - 10.1186/s40792-017-0392-1 [doi] LID - 118 AB - BACKGROUND: Primary hepatic gastrinoma causing severe ulcerogenic syndrome is extremely rare. Herein, we report a case of primary hepatic gastrinoma accompanied by hyperplasia of multi-nodular Brunner's glands in a patient who instead, preoperatively, was suspected of having multiple duodenal gastrinomas and hepatic metastasis. CASE PRESENTATION: A 57-year-old woman consulted a clinic complaining of melena, intermittent abdominal pain, diarrhea, and vomiting which had persisted for about 3 years. Six months before her presentation, she underwent segmental resection of the jejunum for acute peritonitis due to the spontaneous jejunal perforation. A blood test revealed that her serum immunoreactive gastrin (IRG) level was 12,037 pg/mL. Subsequently, she was transferred to our hospital. On computed tomography (CT), a hypervascular tumor of 23 mm in the segment 5 (S5) region of the liver was visualized. A selective arterial secretagogue injection test (SASI test) was performed twice. The first SASI test revealed that the hepatic tumor was a gastrinoma, and there was no gastrinoma in the duodeno-pancreatic region. Additionally, somatostatin receptor scintigraphy only visualized the tumor in the liver. However, the second SASI test, which was performed during the administration of a proton pump inhibitor and a somatostatin analog (octreotide acetate), revealed that there may have been gastrinomas existing not only in the liver but also in the upper part of the duodenum or the head of the pancreas. Duodenal endoscopy revealed multiple submucosal tumors in the first and the second portion of the duodenum, although a pathological examination of biopsied specimens obtained from the duodenal lesions was negative for malignant cells. Multiple endocrine neoplasia type 1 (MEN1) was excluded from her family history, and serum levels of both intact parathyroid hormone (iPTH) and calcium were within normal ranges. An anterior segmentectomy of the liver and pancreas-preserving total duodenectomy were performed on September 9, 2013. Postoperatively, her serum immunoreactive gastrin level decreased to less than 50 pg/mL. Pathological study of the resected specimens revealed a gastrinoma in the liver, but no gastrinoma in the duodenum. Interestingly, the duodenal submucosal tumor-like lesions were hyperplastic Brunner's glands. Postoperatively, she has been well without recurrence of hypergastrinemia for 4 years. CONCLUSION: We report a case of primary hepatic gastrinoma in a patient who has been cured for 4 years postoperatively. The diagnosis was somewhat difficult due to the coexisting, multiple hyperplastic Brunner's glands of the duodenum mimicking the submucosal neuroendocrine tumors, which might have developed due to long-term hypergastrinemia. FAU - Hagi, Takaomi AU - Hagi T AD - Department of Gastroenterological Surgery, Kansai Electric Power Hospital, 2-1-7 Fukushima, Fukushima-ku, Osaka, 553-0003, Japan. AD - Department of Gastroenterological Surgery, Osaka University Graduate School of Medicine, Osaka, Japan. FAU - Hosoda, Yohei AU - Hosoda Y AD - Department of Gastroenterological Surgery, Kansai Electric Power Hospital, 2-1-7 Fukushima, Fukushima-ku, Osaka, 553-0003, Japan. FAU - Komoto, Izumi AU - Komoto I AUID- ORCID: 0000-0002-9698-5507 AD - Department of Gastroenterological Surgery, Kansai Electric Power Hospital, 2-1-7 Fukushima, Fukushima-ku, Osaka, 553-0003, Japan. i-komoto@mx.bw.dream.jp. FAU - Uemoto, Shinji AU - Uemoto S AD - Department of Hepato-Biliary-Pancreatic and Transplantation Surgery, Graduate School of Medicine, Kyoto University, Kyoto, Japan. FAU - Hijioka, Susumu AU - Hijioka S AD - Department of Gastroenterology, Aichi Cancer Center Hospital, Nagoya, Japan. FAU - Taki, Yoshiro AU - Taki Y AD - Department of Gastroenterological Surgery, Kansai Electric Power Hospital, 2-1-7 Fukushima, Fukushima-ku, Osaka, 553-0003, Japan. FAU - Nishiyama, Kazuhiro AU - Nishiyama K AD - Department of Gastroenterological Surgery, Kansai Electric Power Hospital, 2-1-7 Fukushima, Fukushima-ku, Osaka, 553-0003, Japan. FAU - Imamura, Masayuki AU - Imamura M AD - Department of Gastroenterological Surgery, Kansai Electric Power Hospital, 2-1-7 Fukushima, Fukushima-ku, Osaka, 553-0003, Japan. LA - eng PT - Journal Article DEP - 20171128 PL - Germany TA - Surg Case Rep JT - Surgical case reports JID - 101662125 PMC - PMC5704026 OTO - NOTNLM OT - Hypergastrinemia OT - Hyperplasia of Brunner's glands OT - Pancreas-preserving total duodenectomy OT - Primary hepatic gastrinoma OT - Selective arterial secretagogue injection test OT - Somatostatin receptor scintigraphy COIS- CONSENT FOR PUBLICATION: Written informed consent was obtained from the patient for the publication of this case report and the accompanying images. COMPETING INTERESTS: The authors declare that they have no competing interests. PUBLISHER'S NOTE: Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations. EDAT- 2017/11/29 06:00 MHDA- 2017/11/29 06:01 PMCR- 2017/11/28 CRDT- 2017/11/29 06:00 PHST- 2017/09/01 00:00 [received] PHST- 2017/11/09 00:00 [accepted] PHST- 2017/11/29 06:00 [entrez] PHST- 2017/11/29 06:00 [pubmed] PHST- 2017/11/29 06:01 [medline] PHST- 2017/11/28 00:00 [pmc-release] AID - 10.1186/s40792-017-0392-1 [pii] AID - 392 [pii] AID - 10.1186/s40792-017-0392-1 [doi] PST - epublish SO - Surg Case Rep. 2017 Nov 28;3(1):118. doi: 10.1186/s40792-017-0392-1.