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1/223. Cushing's syndrome due to ectopic adrenocorticotropic hormone production by a non-metastatic gastrinoma after long-term conservative treatment of zollinger-ellison syndrome.

    This report concerns a case of a Cushing's syndrome 10 years after first diagnosis of a zollinger-ellison syndrome within the same patient. In a 69-year-old female patient symptoms of hypergastrinaemia have been successfully treated with a proton pump inhibitor. Cushing's syndrome was the result of ectopic adrenocorticotropic hormone production by a large cystic gastrin-producing tumour of the pancreatic tail. After resection by subtotal pancreatectomy serum adrenocorticotropic hormone, cortisol, gastrin levels and secretin infusion test returned to normal. In contrast to all other previously published cases of ectopic adrenocorticotropic hormone syndrome associated with zollinger-ellison syndrome, this tumour had not metastasized into the liver and did not show local invasive growth. ( info)

2/223. Surgical resection of intracardiac gastrinoma.

    There has been only one previous report of an intracardiac gastrinoma causing zollinger-ellison syndrome. In this communication we describe the successful surgical resection of a gastrinoma located in the interventricular septum using cardiopulmonary bypass, blood cardioplegia, and mild hypothermia. Preoperative evaluation and histologic examination of the resected tumor strongly suggest that this was a primary intracardiac gastrinoma. ( info)

3/223. A case of zollinger-ellison syndrome produced by gastrinoma in the duodenum accompanied with multiple endocrine neoplasia type 1.

    A case of zollinger-ellison syndrome produced by gastrinoma in the duodenum accompanied by multiple endocrine neoplasia type-1 (MEN-1) is reported. A 46 year-old female underwent distal gastrectomy due to gastric ulcer 5 years ago. As ulceration of the residual stomach recurred, further examination was performed. hyperprolactinemia, hypergastrinemia, primary hyperparathyroidism, pancreatic tumor, and duodenal carcinoid were evident, and the diagnoses of zollinger-ellison syndrome and MEN-1 were established. The origin of the gastrin secretion was suspected to be from the pancreatic tumor, so sampling of the portal blood was performed. As lesion on the gastrinoma in the pancreas could not be identified, total parathyroidectomy was performed for primary hyperparathyroidism. The level of the gastrin secretion, however, remained high. Partial resection of the duodenum for the duodenal carcinoid and a distal pancreatectomy were carried out concurrently. Immunohistochemical study of the anti-gastrin antibody revealed duodenal tumor cells. Initially, the gastrinoma was thought to be in the pancreas, however, the lesion accompanied with MEN-1 and the zollinger-ellison syndrome had occurred in the duodenum. ( info)

4/223. Imaging findings in primary carcinoid tumour of the liver with gastrin production.

    We present a 57-year-old man with zollinger-ellison syndrome who had undergone total gastrectomy 12 years previously. At that time, a cystic mass in the left lobe of the liver was palpated but was not removed. The patient currently had high serum gastrin levels. Abdominal ultrasound, CT and MR images showed a well defined liver mass with solid and cystic components. The lesion was resected and a primary hepatic carcinoid tumour was diagnosed. Post-operatively, serum gastrin levels were normal. A primary liver carcinoid may appear as multicystic liver mass with solid components. Careful exclusion of a primary tumour elsewhere is required to establish the diagnosis of this rare entity. ( info)

5/223. Expression of vesicular monoamine transporters in endocrine hyperplasia and endocrine tumors of the oxyntic stomach.

    BACKGROUND: Gastric enterochromaffin-like (ECL) cells selectively express the vesicular monoamine transporter (VMAT) VMAT2, and enterochromaffin (EC) cells the VMAT1 isoform. Aims: We investigated whether VMAT isoform selection indicates the origin of endocrine hyperplasia and neoplasia from oxyntic ECL or EC cells and may be of prognostic significance in different types of gastric carcinoids. methods: Tissue from patients with chronic atrophic gastritis (CAG), Zollinger-Ellison-syndrome (ZES), gastric carcinoids and neuroendocrine carcinoma (NEC) was investigated by immunohistology and in situ hybridization. RESULTS: endocrine cells forming diffuse, linear, and micronodular hyperplasia in CAG and ZES, as well as oxyntic microcarcinoids expressed both VMAT2 and chromogranin a (CgA) but neither VMAT1 nor serotonin. In five of six sporadic carcinoids VMAT2 and CgA but not VMAT1 were detected. One carcinoid was copositive for VMAT1 and serotonin but negative for VMAT2. Electron microscopy confirmed the VMAT2-positive tumors as ECLoma and the VMAT1-immunoreactive carcinoid as EComa. CONCLUSIONS: VMAT2 and VMAT1 are reliable markers for differentiation of gastric endocrine hyperplasia and neoplasia from ECL and EC cells, respectively. The significance of VMAT2 and VMAT1 as prognostic markers lies in the relatively poor prognosis for EComa compared to ECLoma, characterized by VMAT2 positivity. The absence of both VMAT2 and VMAT1 in NEC may indicate poor prognosis. ( info)

6/223. Mixed ductal-endocrine carcinoma of the pancreas presenting as gastrinoma with zollinger-ellison syndrome: an autopsy case with a 24-year survival period.

    We report an autopsy case of mixed ductal-endocrine carcinoma of the pancreas presenting as gastrinoma with zollinger-ellison syndrome. A 38-year-old Japanese male was found to have zollinger-ellison syndrome and pancreatic gastrinoma, and gastrectomy and resection of the pancreatic tumor were performed. However, hypergastrinemia persisted, and the patient died of disseminated carcinomatosis at 62 years of age, 24 years after the onset of zollinger-ellison syndrome. At autopsy, the main tumor was present in the residual pancreas, and metastases were noted in many organs. In the pancreas and other organs, ductal and endocrine carcinoma areas were mixed and there was a gradual transition between the two. No acinar differentiation was noted. The ductal elements were positive for mucins and carcinoembryonic antigen but negative for neuroendocrine markers, while endocrine elements were positive for chromogranin a and synaptophysin and to a lesser extent for gastrin, but negative for mucins and carcinoembryonic antigen. The ductal elements comprised about 30% of the tumor cells, and endocrine elements 70%. According to the revised world health organization classification, our case was diagnosed as mixed ductal-endocrine carcinoma. Our case is rare because the tumor manifested as gastrinoma with zollinger-ellison syndrome and the patient survived for 24 years. To the best of our knowledge, no such case has been reported. Our case suggests that pancreatic endocrine tumors may evolve into mixed ductal-endocrine carcinomas. ( info)

7/223. Hyperhistaminemia and hyperchlorhydria in basophilic granulocytic leukemia.

    A patient with basophilic granulocytic leukemia had hyperhistaminemia, hyperchlorhydria and gastroduodenal ulceration. Because the patient's whole blood, plasma, and urinary histamine levels were increased and the serum gastrin was normal, it was concluded that endogenous hyperhistaminemia was responsible for an ulcerogenic diathesis clinically indistinguishable from that seen in the zollinger-ellison syndrome. ( info)

8/223. Ovarian mucinous cystadenocarcinoma as a cause of zollinger-ellison syndrome: report of a case and review of the literature.

    Gastrin producing ovarian tumor is a rare cause of the zollinger-ellison syndrome. We report the case of an ovarian carcinoma showing increased plasma gastrin concentration. A 60-yr-old woman presented with epigastric pain and diarrhea. physical examination showed a large mass in the lower abdomen. Computed tomography revealed a large multilocular ovarian cyst. Upper gastrointestinal endoscopy examination showed multiple ulcerations of the stomach and duodenum. The plasma gastrin level was 1500 pg/ml. No tumors were found in the pancreas and duodenum, and salpingo-oophorectomy was performed. Histologic examination revealed a mucinous tumor of borderline malignant potential. Immunoperoxidase studies for gastrin showed many gastrin-producing cells within the epithelium of the tumor. There have been 11 cases (including our patient) of gastrin-producing ovarian tumor reported in the literature. We review here the relevant literature. Although ovarian gastrinoma is extremely rare, it should be considered as a possible cause of the zollinger-ellison syndrome in women. ( info)

9/223. Hypertrophic gastritis, primary diffuse G-cell hyperplasia and pancreatic metaplasia of the gastric mucosa (pseudo-zollinger-ellison syndrome)--case report.

    We report a case of pseudo-zollinger-ellison syndrome in a 17-year-old man presenting with gastrin levels exceeding 2000 pmol/l and BAO 24 mEq/hr. Histologically, apart from hypertrophic gastritis with the thickening of mucosal folds and diffuse G-cell hyperplasia, gastric mucosa was found to contain foci of pancreatic metaplasia. ( info)

10/223. A report of a gastrinoma localized preoperatively by endoscopic ultrasound only and a review of the approach to imaging in zollinger-ellison syndrome.

    The addition of somatostatin receptor scintigraphy and endoscopic ultrasound to the preoperative assessment of patients with zollinger-ellison syndrome has improved the ability to localize gastrinomas. We report a patient with zollinger-ellison syndrome with a gastrinoma localized preoperatively by endoscopic ultrasound only. We review the literature regarding the sensitivity of somatostatin receptor scintigraphy and endoscopic ultrasound and discuss the approach to imaging in zollinger-ellison syndrome. ( info)
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