胃原発末梢性未熟性神経外胚葉性腫瘍peripheral primitive neuroectodermal tumorの1例
末梢性未熟性神経外胚葉性腫瘍は，神経外胚葉性分化を伴う小型円形細胞からなる悪性腫瘍で，小児・若年者の四肢や傍脊柱領域の軟部組織に発生することが多い．今回，我々はまれな胃原発末梢性未熟性神経外胚葉性腫瘍の1例を経験した．症例は68歳の男性で，食欲不振の精査目的に上部消化管内視鏡検査を施行し，噴門部に隆起性病変を指摘された．生検では腫大した核を有する胞体の乏しい小型円形腫瘍細胞の増殖を認めた．免疫組織化学的染色検査を行うも，組織型の特定は困難であった．胃全摘術を施行し，摘出標本での免疫組織化学染色検査でCD99が腫瘍細胞に陽性となり，遺伝子検査でEWS-FLI1融合遺伝子が証明され，末梢性未熟性神...
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| Published in | 日本消化器外科学会雑誌 Vol. 50; no. 11; pp. 872 - 879 |
|---|---|
| Main Authors | , , , , , , , , , , , |
| Format | Journal Article |
| Language | Japanese |
| Published |
一般社団法人 日本消化器外科学会
2017
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| Subjects | |
| Online Access | Get full text |
| ISSN | 0386-9768 1348-9372 |
| DOI | 10.5833/jjgs.2016.0189 |
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| Abstract | 末梢性未熟性神経外胚葉性腫瘍は，神経外胚葉性分化を伴う小型円形細胞からなる悪性腫瘍で，小児・若年者の四肢や傍脊柱領域の軟部組織に発生することが多い．今回，我々はまれな胃原発末梢性未熟性神経外胚葉性腫瘍の1例を経験した．症例は68歳の男性で，食欲不振の精査目的に上部消化管内視鏡検査を施行し，噴門部に隆起性病変を指摘された．生検では腫大した核を有する胞体の乏しい小型円形腫瘍細胞の増殖を認めた．免疫組織化学的染色検査を行うも，組織型の特定は困難であった．胃全摘術を施行し，摘出標本での免疫組織化学染色検査でCD99が腫瘍細胞に陽性となり，遺伝子検査でEWS-FLI1融合遺伝子が証明され，末梢性未熟性神経外胚葉性腫瘍の診断に至った．術後，多発肝転移を来し，4か月目に死亡した．近年，本腫瘍に対する集学的治療の有効性が報告されている．小型円形細胞からなる胃原発腫瘍を認めた際は本疾患を鑑別に挙げ，治療開始前に確定診断を得ることが重要である． |
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| AbstractList | 末梢性未熟性神経外胚葉性腫瘍は，神経外胚葉性分化を伴う小型円形細胞からなる悪性腫瘍で，小児・若年者の四肢や傍脊柱領域の軟部組織に発生することが多い．今回，我々はまれな胃原発末梢性未熟性神経外胚葉性腫瘍の1例を経験した．症例は68歳の男性で，食欲不振の精査目的に上部消化管内視鏡検査を施行し，噴門部に隆起性病変を指摘された．生検では腫大した核を有する胞体の乏しい小型円形腫瘍細胞の増殖を認めた．免疫組織化学的染色検査を行うも，組織型の特定は困難であった．胃全摘術を施行し，摘出標本での免疫組織化学染色検査でCD99が腫瘍細胞に陽性となり，遺伝子検査でEWS-FLI1融合遺伝子が証明され，末梢性未熟性神経外胚葉性腫瘍の診断に至った．術後，多発肝転移を来し，4か月目に死亡した．近年，本腫瘍に対する集学的治療の有効性が報告されている．小型円形細胞からなる胃原発腫瘍を認めた際は本疾患を鑑別に挙げ，治療開始前に確定診断を得ることが重要である． |
| Author | 中村, 敏夫 福井, 康雄 古北, 由仁 寺石, 文則 志摩, 泰生 澁谷, 祐一 西岡, 豊 尾崎, 和秀 岡林, 雄大 高田, 暢夫 松本, 学 岩田, 純 |
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| References | 3) Czekalla R, Fuchs M, Stölzle A, Nerlich A, Poremba C, Schaefer KL, et al. Peripheral primitive neuroectodermal tumor of the stomach in a 14-year-old boy: a case report. Eur J Gastroenterol Hepatol. 2004 Nov;16(12):1391–1400. 7) Inoue M, Wakai T, Korita PV, Sakata J, Kurosaki R, Ogose A, et al. Gastric Ewing sarcoma/primitive neuroectodermal tumor: a case report. Oncol Lett. 2011 Mar;2(2):207–210. 2) Tan Y, Zhang H, Ma GL, Xiao EH, Wang XC. Peripheral primitive neuroectodermal tumor: dynamic CT, MRI and clinicopathological characteristics—analysis of 36 cases and review of the literature. Oncotarget. 2014 Dec;24(5):12968–12977. 15) Virani MJ, Jain S. Primary intraspinal primitive neuroectodermal tumor (PNET): a rare occurrence. Neurol India. 2002 Mar;50(1):75–80. 20) Ladenstein R, Pötschger U, Le Deley MC, Whelan J, Paulussen M, Oberlin O, et al. Primary disseminated multifocal Ewing sarcoma: results of the Euro-EWING 99 trial. J Clin Oncol. 2010 Jul;28(20):3284–3291. 22) Kamata K, Wada R, Yajima N, Sawada M, Wakasa H, Yagihashi S. Primary gastric synovial sarcoma: molecular diagnosis and prediction of prognosis. Clin J Gastroenterol. 2013 Aug;6(4):303–308. 9) Song MJ, An S, Lee SS, Kim BS, Kim J. Primitive neuroectodermal tumor of the stomach: a case report. Int J Surg Pathol. 2016 Sep;24(6):107–110. 17) Gaspar N, Hawkins DS, Dirksen U, Lewis IJ, Ferrari S, Le Deley MC, et al. Ewing sarcoma: current management and future approaches through collaboration. J Clin Oncol. 2015 Sep;33(27):3036–3046. 10) Khuri S, Gilshtein H, Sayidaa S, Bishara B, Kluger Y. Primary Ewing sarcoma/primitive neuroectodermal tumor of the stomach. Case Rep Oncol. 2016 Nov;9(3):666–671. 13) Angervall L, Enzinger FM. Extraskeletal neoplasm resembling Ewing’s sarcoma. Cancer. 1975;36:240–251. 11) Stout AP. A tumor of the ulner nerve. Proc N Y Pathol Soc. 1918;18:2–12. 8) Kim HS, Kim S, Min YD, Kee KH, Hong R. Ewing’s sarcoma of the stomach; rare case of Ewing’s sarcoma and suggestion of new treatment strategy. J Gastric Cancer. 2012 Dec;12(4):258–261. 16) Coffin CM, Dehner LP. Pheripheral neurogenic tumors of the soft tissues in children and adolescents: a clinicopathologic study of 139 cases. Pediatr Pathol. 1989;9:387–407. 12) Ewing J. Diffuse endothelioma of bone. Proc N Y Pathol Soc. 1921;21:17–24. 19) Paulussen M, Craft AW, Lewis I, Douglas C, Dunst J, Schuck A, et al. Results of the EICESS-92 Study: two randomized trials of Ewing’s sarcoma treatment—cyclophosphamide compared with ifosfamide in standard-risk patients and assessment of benefit of etoposide added to standard treatment in high-risk patients. J Clin Oncol. 2008 Sep;26(27):4385–4393. 4) Soulard R, Claude V, Camparo P, Dufau JP, Saint-Blancard P, Gros P. Primitive neuroectodermal tumor of the stomach. Arch Pathol Lab Med. 2005 Jan;129(1):107–110. 5) Colovic RB, Grubor NM, Micev MT, Matic SV, Atkinson HD, Latincic SM. Perigastric extraskeletal Ewing’s sarcoma: a case report. World J Gastroenterol. 2009 Jan;15(2):245–247. 1) 日本整形外科学会 骨軟部腫瘍委員会編．整形外科・病理 悪性軟部腫瘍取扱い規約．第3版．東京：金原出版；2002. p. 155–158. 21) Sahara S, Otsuki Y, Egawa Y, Shimizu S, Yoshizawa Y, Hosoda Y, et al. Primary synovial sarcoma of the stomach—a case report and review of the litarature. Pathol Res Pract. 2013 Nov;209(11):745–750. 23) Abu-Zaid A, Azzam A, Alnajjar A, Al-Hussaini H, Amin T. Desmoplastic small round cell tumor of stomach. Case Rep Gastrointest Med. 2013;2013:907136. 18) Grier HE, Kralio MD, Tarbell NJ, Link MP, Fryer CJ, Pritchard DJ, et al. Addition of ifosfamide and etoposide to standard chemotherapy for Ewing’s sarcoma and primitive neuroectodermal tumor of bone. N Engl J Med. 2003 Feb;348(8):694–701. 24) Doi H, Ichikawa S, Hiraoka A, Ichiryu M, Nakahara H, Ochi H, et al. Primitive neuroectodermal tumor of the pancreas. Intern Med. 2009;48(5):329–333. 6) Rafailidis S, Ballas K, Psarras K, Pavlidis T, Symeonidis N, Marakis G, et al. Primary Ewing sarcoma of the stomach—a newly described entity. Eur Surg Res. 2009 Nov;42(1):17–20. 14) Seemayer TA, Thelmo WL, Bolande RP, Wiglesworth FW. Pheripheral neuroectodermal tumors. Perspect Pediatr Pathol. 1975;2:151. |
| References_xml | – reference: 15) Virani MJ, Jain S. Primary intraspinal primitive neuroectodermal tumor (PNET): a rare occurrence. Neurol India. 2002 Mar;50(1):75–80. – reference: 3) Czekalla R, Fuchs M, Stölzle A, Nerlich A, Poremba C, Schaefer KL, et al. Peripheral primitive neuroectodermal tumor of the stomach in a 14-year-old boy: a case report. Eur J Gastroenterol Hepatol. 2004 Nov;16(12):1391–1400. – reference: 13) Angervall L, Enzinger FM. Extraskeletal neoplasm resembling Ewing’s sarcoma. Cancer. 1975;36:240–251. – reference: 16) Coffin CM, Dehner LP. Pheripheral neurogenic tumors of the soft tissues in children and adolescents: a clinicopathologic study of 139 cases. Pediatr Pathol. 1989;9:387–407. – reference: 19) Paulussen M, Craft AW, Lewis I, Douglas C, Dunst J, Schuck A, et al. Results of the EICESS-92 Study: two randomized trials of Ewing’s sarcoma treatment—cyclophosphamide compared with ifosfamide in standard-risk patients and assessment of benefit of etoposide added to standard treatment in high-risk patients. J Clin Oncol. 2008 Sep;26(27):4385–4393. – reference: 23) Abu-Zaid A, Azzam A, Alnajjar A, Al-Hussaini H, Amin T. Desmoplastic small round cell tumor of stomach. Case Rep Gastrointest Med. 2013;2013:907136. – reference: 9) Song MJ, An S, Lee SS, Kim BS, Kim J. Primitive neuroectodermal tumor of the stomach: a case report. Int J Surg Pathol. 2016 Sep;24(6):107–110. – reference: 24) Doi H, Ichikawa S, Hiraoka A, Ichiryu M, Nakahara H, Ochi H, et al. Primitive neuroectodermal tumor of the pancreas. Intern Med. 2009;48(5):329–333. – reference: 2) Tan Y, Zhang H, Ma GL, Xiao EH, Wang XC. Peripheral primitive neuroectodermal tumor: dynamic CT, MRI and clinicopathological characteristics—analysis of 36 cases and review of the literature. Oncotarget. 2014 Dec;24(5):12968–12977. – reference: 17) Gaspar N, Hawkins DS, Dirksen U, Lewis IJ, Ferrari S, Le Deley MC, et al. Ewing sarcoma: current management and future approaches through collaboration. J Clin Oncol. 2015 Sep;33(27):3036–3046. – reference: 12) Ewing J. Diffuse endothelioma of bone. Proc N Y Pathol Soc. 1921;21:17–24. – reference: 20) Ladenstein R, Pötschger U, Le Deley MC, Whelan J, Paulussen M, Oberlin O, et al. Primary disseminated multifocal Ewing sarcoma: results of the Euro-EWING 99 trial. J Clin Oncol. 2010 Jul;28(20):3284–3291. – reference: 6) Rafailidis S, Ballas K, Psarras K, Pavlidis T, Symeonidis N, Marakis G, et al. Primary Ewing sarcoma of the stomach—a newly described entity. Eur Surg Res. 2009 Nov;42(1):17–20. – reference: 21) Sahara S, Otsuki Y, Egawa Y, Shimizu S, Yoshizawa Y, Hosoda Y, et al. Primary synovial sarcoma of the stomach—a case report and review of the litarature. Pathol Res Pract. 2013 Nov;209(11):745–750. – reference: 4) Soulard R, Claude V, Camparo P, Dufau JP, Saint-Blancard P, Gros P. Primitive neuroectodermal tumor of the stomach. Arch Pathol Lab Med. 2005 Jan;129(1):107–110. – reference: 10) Khuri S, Gilshtein H, Sayidaa S, Bishara B, Kluger Y. Primary Ewing sarcoma/primitive neuroectodermal tumor of the stomach. Case Rep Oncol. 2016 Nov;9(3):666–671. – reference: 22) Kamata K, Wada R, Yajima N, Sawada M, Wakasa H, Yagihashi S. Primary gastric synovial sarcoma: molecular diagnosis and prediction of prognosis. Clin J Gastroenterol. 2013 Aug;6(4):303–308. – reference: 14) Seemayer TA, Thelmo WL, Bolande RP, Wiglesworth FW. Pheripheral neuroectodermal tumors. Perspect Pediatr Pathol. 1975;2:151. – reference: 1) 日本整形外科学会 骨軟部腫瘍委員会編．整形外科・病理 悪性軟部腫瘍取扱い規約．第3版．東京：金原出版；2002. p. 155–158. – reference: 5) Colovic RB, Grubor NM, Micev MT, Matic SV, Atkinson HD, Latincic SM. Perigastric extraskeletal Ewing’s sarcoma: a case report. World J Gastroenterol. 2009 Jan;15(2):245–247. – reference: 11) Stout AP. A tumor of the ulner nerve. Proc N Y Pathol Soc. 1918;18:2–12. – reference: 7) Inoue M, Wakai T, Korita PV, Sakata J, Kurosaki R, Ogose A, et al. Gastric Ewing sarcoma/primitive neuroectodermal tumor: a case report. Oncol Lett. 2011 Mar;2(2):207–210. – reference: 8) Kim HS, Kim S, Min YD, Kee KH, Hong R. Ewing’s sarcoma of the stomach; rare case of Ewing’s sarcoma and suggestion of new treatment strategy. J Gastric Cancer. 2012 Dec;12(4):258–261. – reference: 18) Grier HE, Kralio MD, Tarbell NJ, Link MP, Fryer CJ, Pritchard DJ, et al. Addition of ifosfamide and etoposide to standard chemotherapy for Ewing’s sarcoma and primitive neuroectodermal tumor of bone. N Engl J Med. 2003 Feb;348(8):694–701. |
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| Title | 胃原発末梢性未熟性神経外胚葉性腫瘍peripheral primitive neuroectodermal tumorの1例 |
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