Two Cases of Duodenal Somatostatinoma Associated with von Recklinghausen's Disease
We report two cases of duodenal somatostatinoma associated with von Recklinghausen's disease. Case 1 involved a 45-year-old man and case 2 a 67-year-old man, in whom cafe-aulait skin macules and multiple neurofibromas were noted over the entire body. They underwent pancreatoduodenectomy based o...
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| Published in | Nippon Shokaki Geka Gakkai zasshi Vol. 38; no. 8; pp. 1312 - 1317 |
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| Main Authors | , , , , , , , |
| Format | Journal Article |
| Language | Japanese |
| Published |
The Japanese Society of Gastroenterological Surgery
2005
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| Subjects | |
| Online Access | Get full text |
| ISSN | 0386-9768 1348-9372 |
| DOI | 10.5833/jjgs.38.1312 |
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| Abstract | We report two cases of duodenal somatostatinoma associated with von Recklinghausen's disease. Case 1 involved a 45-year-old man and case 2 a 67-year-old man, in whom cafe-aulait skin macules and multiple neurofibromas were noted over the entire body. They underwent pancreatoduodenectomy based on a diagnosis of duodenal carcinoid in the oral side of the papilla of Vater. Histologically, the duodenal tumors in these cases infiltrated the pancreatic head. A high-power view showed tumors to consist of small round cells with a solid or trabecular pattern and conspicuous psammoma bodies. Regional lymph nodes contained metastases. Immunohistochemical examination showed that tumor cells stained for somatostatin and chromogranin. In case 1, ultrastractual study demonstrated neuroendocrine secretory granules (300-400 nm in diameter) in cytoplasm. It is noted that somatostatinoma should be considered on differential diagnosis in patient with von Recklinghausen's disease accompanied with periampullary neoplasm. |
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| AbstractList | We report two cases of duodenal somatostatinoma associated with von Recklinghausen's disease. Case 1 involved a 45-year-old man and case 2 a 67-year-old man, in whom cafe-aulait skin macules and multiple neurofibromas were noted over the entire body. They underwent pancreatoduodenectomy based on a diagnosis of duodenal carcinoid in the oral side of the papilla of Vater. Histologically, the duodenal tumors in these cases infiltrated the pancreatic head. A high-power view showed tumors to consist of small round cells with a solid or trabecular pattern and conspicuous psammoma bodies. Regional lymph nodes contained metastases. Immunohistochemical examination showed that tumor cells stained for somatostatin and chromogranin. In case 1, ultrastractual study demonstrated neuroendocrine secretory granules (300-400 nm in diameter) in cytoplasm. It is noted that somatostatinoma should be considered on differential diagnosis in patient with von Recklinghausen's disease accompanied with periampullary neoplasm. |
| Author | Gonda, Kenzi Nihei, Mitsuhiro Katagata, Naoto Yamaguchi, Yoshiko Nomizu, Tadashi Yamada, Mutsuo Sakuma, Takeshi Watanabe, Fumiaki |
| Author_xml | – sequence: 1 fullname: Nomizu, Tadashi organization: Department of Surgery, Hoshi General Hospital – sequence: 1 fullname: Watanabe, Fumiaki organization: Department of Surgery, Hoshi General Hospital – sequence: 1 fullname: Yamaguchi, Yoshiko organization: Department of Pathology, Hoshi General Hospital – sequence: 1 fullname: Sakuma, Takeshi organization: Department of Surgery, Hoshi General Hospital – sequence: 1 fullname: Yamada, Mutsuo organization: Department of Surgery, Hoshi General Hospital – sequence: 1 fullname: Katagata, Naoto organization: Department of Surgery, Hoshi General Hospital – sequence: 1 fullname: Nihei, Mitsuhiro organization: Igarashi Internal and Surgical Clinic – sequence: 1 fullname: Gonda, Kenzi organization: Department of Surgery, Hoshi General Hospital |
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| References | 13) Dayal Y, Tallberg KA, Nunnemacher G et al: Duodenal carcinoids in patients with and without neurofibromatosis. A comparative study. Am J Surg Pathol 10: 348-357, 1986 3) Kaneko H, Yanaihara N, Ito S et al: Somatostatinoma of the duodenum. Cancer 44: 2273-2279, 1979 14) 中田岳成, 坂井威彦, 熊木俊成ほか: von Recklinghausen病に併存した原発性十二指腸癌の1例. 日消外会誌 34: 1611-1615, 2001 1) 日本胆道外科研究会編: 胆道癌取扱い規約. 第4版. 金原出版, 東京, 1997 4) Soga J, Yakuwa Y: Somatostatinoma/Inhibitory syndrome: a statistical evaluation of 173 reported cases as compared to other pancreatic endocrinomas. J Exp Clin Cancer Res 18: 13-22, 1999 8) Fuller CE, Williams GT: Gastrointestinal manifestations of type 1 neurofibromatosis (von Recklinghausen's disease). Histopathology 19: 1-11, 1991 17) O'Brien TD, Chejfec G, Prinz RA: Clinical features of duodenal somatostatinomas. Surgery 114: 1144-1147, 1993 12) Xu GF, O'Connell P, Viskochil D et al: The neurofibromatosis type 1 gene encodes a protein related to GAP. Cell 62: 599-608, 1990 9) Mao C, Shah A, Hanson DJ et al: Von Reckling hausen's disease associated with duodenal somatostatinoma: contrast of duodenal versus pancreatic somatostatinomas. J Surg Oncol 59: 67-73, 1995 10) Krejs GJ, Orci L, Conlon JM et al: Somatostatinoma syndrome. Biochemical, morphological and clinical features. N Engl J Med 301: 285-292, 1979 2) Larsson LI, Hirsh MA, Holst JJ et al: Pancreatic somatostatinoma. Clinical features and physiological implications. Lancet 1: 666-668, 1977 5) Stommer PE, Stolte M, Seifert E: Somatostatinoma of Vater's papilla and of the minor papilla. Cancer 60: 232-235, 1987 11) Martin GA, Viskochil D, Bollag G et al: The GA-P-related domain of the neurofibromatosis type 1 gene product interacts with ras p 21. Cell 63: 843-849, 1990 15) Tanaka S, Yamasaki S, Matsushita H et al: Duodenal somatostatinoma: a case report and review of 31 cases with special reference to the relationship between tumor size and metastasis. Pathol Int 50: 146-152, 2000 16) Pipeleers D, Somers G, Gepts W et al: Plasma pancreatic hormone levels in a case of somatostatinoma: diagnostic and therapeutic implications. J Clin Endocrinol Metab 49: 572-579, 1979 6) Albores-Saavedra J, Henson DE, Klimstra DS: Tumors of the gallbladder, extrahepatic bile ducts. and ampulla of vater. Armed forces institute of pathology. Washington, DC, 2000, p321-325 7) Capella C, Solcia E, Sobin LH et al: Endocrine tumors of the small intestine. Edited by Hamilton SR, Aaltonen LA. Pathology and genetics of the digestive system: WHO Classification of Tumors. IARC Press, Lyon, 2000, p77-82 |
| References_xml | – reference: 13) Dayal Y, Tallberg KA, Nunnemacher G et al: Duodenal carcinoids in patients with and without neurofibromatosis. A comparative study. Am J Surg Pathol 10: 348-357, 1986 – reference: 11) Martin GA, Viskochil D, Bollag G et al: The GA-P-related domain of the neurofibromatosis type 1 gene product interacts with ras p 21. Cell 63: 843-849, 1990 – reference: 6) Albores-Saavedra J, Henson DE, Klimstra DS: Tumors of the gallbladder, extrahepatic bile ducts. and ampulla of vater. Armed forces institute of pathology. Washington, DC, 2000, p321-325 – reference: 15) Tanaka S, Yamasaki S, Matsushita H et al: Duodenal somatostatinoma: a case report and review of 31 cases with special reference to the relationship between tumor size and metastasis. Pathol Int 50: 146-152, 2000 – reference: 16) Pipeleers D, Somers G, Gepts W et al: Plasma pancreatic hormone levels in a case of somatostatinoma: diagnostic and therapeutic implications. J Clin Endocrinol Metab 49: 572-579, 1979 – reference: 10) Krejs GJ, Orci L, Conlon JM et al: Somatostatinoma syndrome. Biochemical, morphological and clinical features. N Engl J Med 301: 285-292, 1979 – reference: 1) 日本胆道外科研究会編: 胆道癌取扱い規約. 第4版. 金原出版, 東京, 1997 – reference: 14) 中田岳成, 坂井威彦, 熊木俊成ほか: von Recklinghausen病に併存した原発性十二指腸癌の1例. 日消外会誌 34: 1611-1615, 2001 – reference: 3) Kaneko H, Yanaihara N, Ito S et al: Somatostatinoma of the duodenum. Cancer 44: 2273-2279, 1979 – reference: 4) Soga J, Yakuwa Y: Somatostatinoma/Inhibitory syndrome: a statistical evaluation of 173 reported cases as compared to other pancreatic endocrinomas. J Exp Clin Cancer Res 18: 13-22, 1999 – reference: 7) Capella C, Solcia E, Sobin LH et al: Endocrine tumors of the small intestine. Edited by Hamilton SR, Aaltonen LA. Pathology and genetics of the digestive system: WHO Classification of Tumors. IARC Press, Lyon, 2000, p77-82 – reference: 9) Mao C, Shah A, Hanson DJ et al: Von Reckling hausen's disease associated with duodenal somatostatinoma: contrast of duodenal versus pancreatic somatostatinomas. J Surg Oncol 59: 67-73, 1995 – reference: 2) Larsson LI, Hirsh MA, Holst JJ et al: Pancreatic somatostatinoma. Clinical features and physiological implications. Lancet 1: 666-668, 1977 – reference: 5) Stommer PE, Stolte M, Seifert E: Somatostatinoma of Vater's papilla and of the minor papilla. Cancer 60: 232-235, 1987 – reference: 12) Xu GF, O'Connell P, Viskochil D et al: The neurofibromatosis type 1 gene encodes a protein related to GAP. Cell 62: 599-608, 1990 – reference: 8) Fuller CE, Williams GT: Gastrointestinal manifestations of type 1 neurofibromatosis (von Recklinghausen's disease). Histopathology 19: 1-11, 1991 – reference: 17) O'Brien TD, Chejfec G, Prinz RA: Clinical features of duodenal somatostatinomas. Surgery 114: 1144-1147, 1993 |
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| Title | Two Cases of Duodenal Somatostatinoma Associated with von Recklinghausen's Disease |
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