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Sugimoto A, Nishida T, Hosokawa K, Fujii Y, Nakamatsu D, Matsumoto K, Yamamoto M, Fukui K. Primary Neuroendocrine Carcinoma of the Ileum With Markedly Elevated Carcinoembryonic Antigen (CEA) Levels: A Case Report. Cureus 2024; 16:e66676. [PMID: 39262550 PMCID: PMC11389073 DOI: 10.7759/cureus.66676] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 08/12/2024] [Indexed: 09/13/2024] Open
Abstract
Neuroendocrine carcinomas (NECs) are rare and highly malignant tumors with a generally poor prognosis. Carcinoembryonic antigen (CEA) is often associated with adenocarcinoma, but its significant elevation in NEC cases is unusual. A 69-year-old man was admitted to our hospital in January 2016 due to syncope induced by anemia. The patient had a hemoglobin level of 8.0 g/dL and an ileocecal mass causing small bowel obstruction on computed tomography. His CEA level was markedly elevated at 3625.4 ng/mL. A colonoscopy revealed a neoplastic lesion in the terminal ileum, leading to an emergency ileocecal resection. Pathology confirmed a NEC, positive for synaptophysin and CEA, with a Ki-67 index of 30%. The patient was diagnosed with stage IIIb NEC (pT3N2M0). A postoperative increase in CEA to 4124.6 ng/mL and metastases in the right lung and multiple lymph nodes were detected. Initial chemotherapy with irinotecan, cisplatin (IP), and octreotide acetate proved ineffective. Subsequent octreoscans showed disease progression. Switching to everolimus as second-line therapy temporarily decreased CEA levels and tumor size, but the disease progressed with cervical lymph node involvement. The patient underwent palliative radiotherapy but succumbed to disease progression in May 2018, with a final CEA level of 36,643 ng/mL. Necropsy of the cervical lymph nodes was consistent with the original surgical findings. This case highlights the aggressive nature and challenging management of NEC with significantly elevated CEA levels.
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Affiliation(s)
- Aya Sugimoto
- Department of Gastroenterology, Toyonaka Municipal Hospital, Toyonaka, JPN
| | - Tsutomu Nishida
- Department of Gastroenterology, Toyonaka Municipal Hospital, Toyonaka, JPN
| | - Kana Hosokawa
- Department of Gastroenterology, Toyonaka Municipal Hospital, Toyonaka, JPN
| | - Yoshifumi Fujii
- Department of Gastroenterology, Toyonaka Municipal Hospital, Toyonaka, JPN
| | - Dai Nakamatsu
- Department of Gastroenterology, Toyonaka Municipal Hospital, Toyonaka, JPN
| | - Kengo Matsumoto
- Department of Gastroenterology, Toyonaka Municipal Hospital, Toyonaka, JPN
| | - Masashi Yamamoto
- Department of Gastroenterology, Toyonaka Municipal Hospital, Toyonaka, JPN
| | - Koji Fukui
- Department of Gastroenterology, Toyonaka Municipal Hospital, Toyonaka, JPN
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Assis AC, Tercioti V, Andreollo NA, Ferrer JAP, Coelho JDS, Lopes LR. GASTRIC NEUROENDOCRINE TUMOR: WHEN SURGICAL TREATMENT IS INDICATED? ARQUIVOS BRASILEIROS DE CIRURGIA DIGESTIVA : ABCD = BRAZILIAN ARCHIVES OF DIGESTIVE SURGERY 2023; 36:e1768. [PMID: 37851754 PMCID: PMC10578147 DOI: 10.1590/0102-672020230050e1768] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 12/13/2022] [Accepted: 08/17/2023] [Indexed: 10/20/2023]
Abstract
BACKGROUND Gastric neuroendocrine tumors are a heterogeneous group of neoplasms that produce bioactive substances. Their treatment varies according to staging and classification, using endoscopic techniques, open surgery, chemotherapy, radiotherapy, and drugs analogous to somatostatin. AIMS To identify and review cases of gastric neuroendocrine neoplasia submitted to surgical treatment. METHODS Review of surgically treated patients from 1983 to 2018. RESULTS Fifteen patients were included, predominantly female (73.33%), with a mean age of 55.93 years. The most common symptom was epigastric pain (93.3%), and the mean time of symptom onset was 10.07 months. The preoperative upper digestive endoscopy (UDE) indicated a predominance of cases with 0 to 1 lesion (60%), sizing ≥1.5 cm (40%), located in the gastric antrum (53.33%), with ulceration (60%), and Borrmann III (33.33%) classification. The assessment of the surgical specimen indicated a predominance of invasive neuroendocrine tumors (60%), with angiolymphatic invasion in most cases (80%). Immunohistochemistry for chromogranin A was positive in 60% of cases and for synaptophysin in 66.7%, with a predominant Ki-67 index between 0 and 2%. Metastasis was observed in 20% of patients. The surgical procedure most performed was subtotal gastrectomy with Roux-en-Y reconstruction (53.3%). Tumor recurrence occurred in 20% of cases and a new treatment was required in 26.67%. CONCLUSIONS Gastric neuroendocrine tumors have a low incidence in the general population, and surgical treatment is indicated for advanced lesions. The study of its management gains importance in view of the specificities of each case and the need for adequate conduct to prevent recurrences and complications.
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Affiliation(s)
| | - Valdir Tercioti
- Universidade Estadual de Campinas, Faculty of Medical Sciences, Department of Surgery and Gastro Center – Campinas (SP), Brazil
| | - Nelson Adami Andreollo
- Universidade Estadual de Campinas, Faculty of Medical Sciences, Department of Surgery and Gastro Center – Campinas (SP), Brazil
| | - José Antonio Possatto Ferrer
- Universidade Estadual de Campinas, Faculty of Medical Sciences, Department of Surgery and Gastro Center – Campinas (SP), Brazil
| | - João de Souza Coelho
- Universidade Estadual de Campinas, Faculty of Medical Sciences, Department of Surgery and Gastro Center – Campinas (SP), Brazil
| | - Luiz Roberto Lopes
- Universidade Estadual de Campinas, Faculty of Medical Sciences, Department of Surgery and Gastro Center – Campinas (SP), Brazil
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Boeriu A, Dobru D, Fofiu C, Brusnic O, Onişor D, Mocan S. Gastric neuroendocrine neoplasms and precursor lesions: Case reports and literature review. Medicine (Baltimore) 2022; 101:e28550. [PMID: 35029217 PMCID: PMC8757942 DOI: 10.1097/md.0000000000028550] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/06/2021] [Accepted: 12/22/2021] [Indexed: 02/07/2023] Open
Abstract
RATIONALE Gastric neuroendocrine neoplasms (g-NENs) represent a distinctive group of gastric tumors, stratified into different prognostic categories according to different histological characteristics, put forth in the 2018 World Health Organization classification system. The clinical presentations, as well as pathological features, represent important data in establishing the type of the tumor, in estimating the tumor behavior, and in selecting the best therapeutic strategy. In our case series we presented different clinical scenarios that may be encountered in practice regarding gastric NENs. We performed a literature review and discussed diagnostic strategy, current classification system, precursor lesions, and therapeutic options in g-NENs. PATIENT CONCERNS The first patient was a 41-year-old female with weight loss, persistent dyspeptic complaints and a history of pernicious anemia. In the second clinical case a 61-year-old man was admitted with heartburn, abdominal pain, diarrhea and mild iron deficiency anemia. The third patient was a 56-year-old male with a history of neoplasia, admitted for weight loss, dyspeptic complaints, and liver metastases. DIAGNOSIS All the 3 patients underwent upper endoscopy with targeted biopsies. Histopathological and laboratory evaluation, together with imagistic evaluation (abdominal ultrasound, endoscopic ultrasound, and magnetic resonance imaging) allowed the distinction between 3 different types of gastric tumors: type 1 enterochromaffin-like-cell G1 NET, type 2 enterochromaffin-like-cell G2 NET, and type 3 G2 NET with liver metastases. INTERVENTIONS Endoscopic polypectomy of the largest lesion was performed in patient with type 1 g-NET and autoimmune chronic atrophic gastritis, followed by regular endoscopic surveillance with biopsies. In type 2 g-NET associated with pancreatic gastrinoma, pancreaticoduodenectomy with total gastrectomy were performed. In type 3 g-NET, detected in metastatic stage, oncologic therapy was performed. OUTCOMES The patients follow-up was selected according to tumor behavior, from regular endoscopic surveillance to oncology follow-up. The prognosis was good in case 1, whilst poorer outcomes were associated with more aggressive tumors in case 2 and case 3. LESSONS g-NENs are rare tumors with distinct clinical and histological features. Our case series emphasized the role of close collaboration between clinician and pathologist, as well as the importance of a detailed pathology report.
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Affiliation(s)
- Alina Boeriu
- Department of Gastroenterology, University of Medicine, Pharmacy, Science, and Technology of Targu Mures, Romania
| | - Daniela Dobru
- Department of Gastroenterology, University of Medicine, Pharmacy, Science, and Technology of Targu Mures, Romania
| | - Crina Fofiu
- Department of Gastroenterology, University of Medicine, Pharmacy, Science, and Technology of Targu Mures, Romania
| | - Olga Brusnic
- Department of Gastroenterology, University of Medicine, Pharmacy, Science, and Technology of Targu Mures, Romania
| | - Danusia Onişor
- Department of Gastroenterology, University of Medicine, Pharmacy, Science, and Technology of Targu Mures, Romania
| | - Simona Mocan
- Pathology Department, University of Medicine, Pharmacy, Science, and Technology of Targu Mures, Romania
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Bellorin O, Shuchleib A, Halevi AE, Aksenov S, Saldinger PF. Giant type III well-differentiated neuroendocrine tumor of the stomach: A case report. Int J Surg Case Rep 2016; 25:62-5. [PMID: 27327559 PMCID: PMC4917394 DOI: 10.1016/j.ijscr.2016.06.008] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/03/2016] [Revised: 06/09/2016] [Accepted: 06/09/2016] [Indexed: 12/28/2022] Open
Abstract
INTRODUCTION We describe a case of a large type III neuroendocrine tumor of the stomach. Management and current literature are reviewed. PRESENTATION OF CASE A 37year old female presented with upper gastrointestinal bleed and epigastric pain. Further workup demonstrated a large ulcerated gastric mass near the GE junction. Computer tomography scan and endoscopic ultrasound showed a 10cm mass with no evidence of distant disease. Fine needle aspiration pathology was consistent with a well differentiated neuroendocrine tumor (Ki67 index<2%), with elevated levels of chromogranin A and serotonin levels but normal gastrin. The patient underwent an uneventful total gastrectomy. Final pathology analysis reported a higher KI67 index (7.54%) and a final pathology of grade 2 type III, T3 N3, neuroendocrine tumor of the stomach. The chromogranin levels normalized and no recurrent disease has been detected in one year follow up. DISCUSSION Gastric neuroendocrine tumors are extremely rare, accounting for 4% of all neuroendocrine tumors of the body and 1% of all neoplasms of the stomach. Based on histomorphologic characteristics and pathogenesis, gastric neuroendocrine tumors are classified into four types with differing prognosis and behavior. Current literature describes type 3 gastric neuroendocrine tumors as larger than 2cm. However, there is no precedent in the literature for a tumor of this size. CONCLUSION The incidence of gastric neuroendocrine tumors has been increasing during the last decade, underscoring the need to improve our understanding of their biology and behavior. When identified histologically, patient outcomes depend on appropriate determination of tumor biology and subsequent choice of treatment.
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Affiliation(s)
- Omar Bellorin
- Department of General Surgery, New York Presbyterian Queens/Weill Cornell Medical College, 5645 Main Street, suite 300, Flushing, NY 11355, USA.
| | - Ariel Shuchleib
- Department of General Surgery, New York Presbyterian Queens/Weill Cornell Medical College, 5645 Main Street, suite 300, Flushing, NY 11355, USA.
| | - Alexandra E Halevi
- Department of General Surgery, New York Presbyterian Queens/Weill Cornell Medical College, 5645 Main Street, suite 300, Flushing, NY 11355, USA.
| | - Sergei Aksenov
- Department of Pathology, New York Presbyterian Queens/Weill Cornell Medical College, 5645 Main Street, Flushing, NY 11355, USA.
| | - Pierre F Saldinger
- Department of General Surgery, New York Presbyterian Queens/Weill Cornell Medical College, 5645 Main Street, suite 300, Flushing, NY 11355, USA.
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Funahashi H, Miyai H, Wakasugi T, Ishiguro H, Matsuo Y, Kimura M, Takeyama H. Successful combination chemotherapy with irinotecan hydrochloride and cisplatin for primary gastric small cell carcinoma: report of a case. World J Surg Oncol 2013; 11:263. [PMID: 24099520 PMCID: PMC3852596 DOI: 10.1186/1477-7819-11-263] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/25/2013] [Accepted: 09/27/2013] [Indexed: 12/29/2022] Open
Abstract
Primary gastric small cell carcinoma is a rare and aggressive malignant disease with a poor prognosis that was first reported in 1976 by Matsusaka et al. The incidence is very low and the clinicopathological features are similar to those of small cell lung carcinoma. We herein report a case of successful treatment by combination chemotherapy consisting of irinotecan hydrochloride and cisplatin for primary gastric small cell carcinoma. The patient was a 71-year-old male who was admitted to a local hospital with anemia. Gastrointestinal endoscopy revealed the presence of advanced gastric carcinoma at the upper region of the stomach. The patient underwent surgery, and the pathological diagnosis was small cell carcinoma due to the presence of the typical features of small round cells with scant cytoplasm that were positive for synaptophysin and chromogranin A in the resected specimen. The patient underwent subsequent combination chemotherapy, which provided him with over 1 year of survival and a good quality of life. We also present a review of the literature regarding chemotherapy for primary gastric small cell carcinoma.
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Affiliation(s)
- Hitoshi Funahashi
- Department of Gastroenterological Surgery, Nagoya City University Graduate School of Medical Sciences, 1 Kawasumi, Mizuho-cho, Mizuho-ku, Nagoya 4678601, Japan.
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Lin BS. MSCT manifestations and pathological features of gastric tumors: An analysis of 56 cases. Shijie Huaren Xiaohua Zazhi 2013; 21:915-919. [DOI: 10.11569/wcjd.v21.i10.915] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/06/2023] Open
Abstract
AIM: To analyze the multi-slice spiral computed tomography (MSCT) features of different pathological types of gastric tumors to improve their diagnosis and differential diagnosis.
METHODS: Imaging and pathological data for 56 patients with gastric tumors who underwent MSCT dual-phase enhanced scan were reviewed retrospectively. The MSCT features and enhancement characteristics were compared among different pathological types of gastric tumors.
RESULTS: The pathological types of 56 cases of gastric tumors included adenocarcinoma (n = 48), mesenchymal tumor (n = 4), lymphoma (n = 2), neuroendocrine carcinoma (n = 1), and hepatoid adenocarcinoma (n = 1). Mesenchymal tumors showed specific growth pattern. MSCT enhancement manifestations of various types of gastric tumors were similar.
CONCLUSION: Observation of tumor growth pattern and enhancement manifestations by MSCT allows for improvement of the diagnostic accuracy of gastric tumors.
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Kuo SC, Chao Y, Luo JC, Lee KC, Wu CW, Li AFY, Lee RC, Li CP. Primary small cell carcinoma of the stomach successfully treated with cisplatin and etoposide. J Chin Med Assoc 2009; 72:598-602. [PMID: 19948438 DOI: 10.1016/s1726-4901(09)70436-8] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 10/20/2022] Open
Abstract
We report a 44-year-old man with primary gastric small cell carcinoma who showed a remarkable response to chemotherapy specific for pulmonary small cell carcinoma. The patient had been admitted to another local hospital because of intermittent epigastralgia. An upper gastrointestinal examination there revealed an ulcerative tumor, 5 cm in diameter, on the lesser curvature side of the cardia, and endoscopic biopsy reported adenocarcinoma. Computed tomography revealed a mass over the lesser curvature of the stomach and some enlarged regional lymph nodes. Radical total gastrectomy, lymph node dissection, Roux-en-Y esophagojejunostomy and splenectomy were performed at our hospital. Pathology revealed gastric mucosa infiltrated by small-sized tumor cells with scanty cytoplasm and hyperchromatic nuclei. Immunohistochemically, the tumor cells were positive for synaptophysin, chromogranin A, and CD56. Primary gastric small cell carcinoma was diagnosed. The postoperative course, complicated by shock due to bleeding, wound infection and intra-abdominal abscess, took more than 2 months to resolve. Follow-up computed tomography showed tumor recurrence with multiple enlarged lymph nodes in the aortocaval region and hepatic hilum. The patient received palliative chemotherapy consisting of cisplatin 80 mg/m(2) on day 1 and etoposide 80 mg/m(2) on days 1-3 every 28 days, and had partial response to the chemotherapy, with a progression-free survival of 10 months. Chemotherapy with cisplatin and etoposide used for small cell carcinoma of the lung is a good treatment for gastric small cell carcinoma.
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Affiliation(s)
- Shu-Chen Kuo
- Division of Gastroenterology, Department of Medicine, Taipei Veterans General Hospital, Taipei, Taiwan, R.O.C
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Waisberg J, de Matos LL, do Amaral Antonio Mader AM, Pezzolo S, Eher EM, Capelozzi VL, Speranzini MB. Neuroendocrine gastric carcinoma expressing somatostatin: A highly malignant, rare tumor. World J Gastroenterol 2006; 12:3944-7. [PMID: 16804989 PMCID: PMC4087952 DOI: 10.3748/wjg.v12.i24.3944] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/06/2023] Open
Abstract
Poorly differentiated gastric neuroendocrine carcinomas, although rare, deserve particular attention, as they are aggressive and have an extremely poor prognosis. In this report we describe a gastric neuroendocrine carcinoma with rapidly fatal outcome. Immunohistological staining of the resected specimens revealed that the tumor was an endocrine carcinoma. The tumor disclosed intense immunoreactivity to pan-neuroendocrine markers and diffuse somatostatin immunoreactivity. There were no psammoma bodies and no demonstrable association with von Recklinghausen’s neurofibromatosis. In the gastrointestinal tract, neuroendocrine tumors producing predominantly somatostatin have been described only in the duodenum. To the best of our knowledge, the present report is the second case report of a neuroendocrine gastric carcinoma expressing diffusely somatostatin as the only neuroendocrine regulatory peptide.
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Affiliation(s)
- Jaques Waisberg
- Department of Surgery, Faculty of Medicine of ABC, Santo André, Braz.
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Goldstein NS, Bosler DS. Immunohistochemistry of the Gastrointestinal Tract, Pancreas, Bile Ducts, Gallbladder and Liver. DIAGNOSTIC IMMUNOHISTOCHEMISTRY 2006:442-508. [DOI: 10.1016/b978-0-443-06652-8.50019-3] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/05/2025]
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Nakamura Y, Otani S, Otaka M, Shimada T, Takahashi S, Saito M, Takahashi T, Komatsu M, Suzuki T, Okubo S, Hayashi M, Sasano H. Gastric small cell carcinoma with marked response to neoadjuvant chemotherapy. Int J Clin Oncol 2005; 10:348-52. [PMID: 16247663 DOI: 10.1007/s10147-005-0492-y] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/14/2004] [Accepted: 03/30/2005] [Indexed: 10/25/2022]
Abstract
We report a patient with gastric small cell carcinoma (SCC) who showed a marked response to neoadjuvant chemotherapy. The patient was a 72-year-old Japanese man who was admitted because of epigastralgia. Subsequent examination revealed the presence of advanced gastric carcinoma in the lesser curvature of the lower body of the stomach, with multiple abdominal lymph node metastases. Endoscopic biopsy specimens from the tumor revealed SCC with moderately differentiated adenocarcinoma. The patient received neoadjuvant chemotherapy consisting of carboplatin (400 mg/m2, for 1 day), epirubicin (27 mg/m2, for 1 day), etoposide (70 mg/m2, for 3 days), and 5-fluorouracil (330 mg/m2, for 11 days). Clinically, the primary tumor and lymph node metastases were markedly reduced. Subsequently the patient underwent curative surgery and was alive without recurrence for more than 3 years after the surgery.
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Affiliation(s)
- Yasuhiro Nakamura
- Department of Pathology, Tohoku Graduate University School of Medicine, 2-1 Seiryo-machi, Sendai 980-8575, Japan.
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Fujii T, Kawai T, Saito K, Hishima T, Hayashi Y, Imura J, Hironaka M, Hosoya Y, Koike M, Fukayama M. MEN1 gene mutations in sporadic neuroendocrine tumors of foregut derivation. Pathol Int 1999; 49:968-73. [PMID: 10594843 DOI: 10.1046/j.1440-1827.1999.00971.x] [Citation(s) in RCA: 23] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/20/2022]
Abstract
Foregut-derived neuroendocrine (NE) tumors occur sporadically or in association with multiple endocrine neoplasia type 1 (MEN1) syndrome. Thirty-nine sporadic NE tumors of foregut derivation (six thymic, 21 bronchial, three gastric, and nine pancreatic tumors) as well as two hindgut-derived rectal carcinoids for somatic MEN1 gene mutation were analyzed by direct sequencing analysis. Five tumors showed mutations: nonsense mutations (Q393X and R98X) in thymic and pancreatic NE tumors, respectively, a 4 b.p. deletion (357del4) in a gastric NE carcinoma, and missense mutations (D172Y and S178Y) in pancreatic NE tumors. No mutation was identified in pulmonary or rectal NE tumors. In a patient with a pancreatic NE tumor (D172Y), the corresponding germline DNA showed the same mutation, suggesting that sporadic MEN1 syndrome was masked in this case. Somatic MEN1 gene mutations and deletions may play a crucial role in the tumorigenesis of a subset of foregut-derived NE tumors. Sporadic MEN1 syndrome may occur as a sporadic NE tumor of the pancreas.
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Affiliation(s)
- T Fujii
- Department of Pathology, Jichi Medical School, Minamikawachi, Kawachi, Tochigi, Japan.
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