MDM2遺伝子増幅を認めた顎下部異型脂肪腫様腫瘍の1例

Atypical lipomatous tumor (ALT)/well-differentiated liposarcoma, a relatively common malignant soft-tissue tumor, was first described in 1857 by Rudolf Virchow. It generally occurs in the four limbs and the retroperitoneum and rarely develops in the head and neck region. Here we report a case of an...

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Published in日本口腔外科学会雑誌 Vol. 66; no. 10; pp. 483 - 490
Main Authors 福村, 元洋, 鈴木, 慎太郎, 長尾, 徹, 後藤, 大輝, 森下, 佳学, 宮部, 悟
Format Journal Article
LanguageJapanese
Published 社団法人 日本口腔外科学会 20.10.2020
Subjects
MDM2増幅
異型脂肪腫様腫瘍
顎下部
Online AccessGet full text
ISSN0021-5163
2186-1579
DOI10.5794/jjoms.66.483

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Abstract Atypical lipomatous tumor (ALT)/well-differentiated liposarcoma, a relatively common malignant soft-tissue tumor, was first described in 1857 by Rudolf Virchow. It generally occurs in the four limbs and the retroperitoneum and rarely develops in the head and neck region. Here we report a case of an atypical lipomatous tumor in the left submandibular region. A 57-year-old man presented with a slowly enlarging mass in the lower left submandibular region. Initial examinations using computed tomography and magnetic resonance imaging revealed heterogeneous internal patterns; therefore, we performed an open biopsy. Histological examination of the specimen indicated lipoma. In February 2011, the patient underwent tumor excision under general anesthesia. The tumor adhered to the lingual nerve; we divided the tumor into parts to save the nerves and resected as much of the tumor as possible. Based on the histological examination of the resected specimens, an atypical lipomatous tumor was diagnosed. We re-operated in April to excise the remainder of the tumor. We excised it along with the lingual nerve via a mandibular swing approach because the tumor was in contact with the medial pterygoid muscle in the medial aspect of the mandible. No signs of recurrence were noted after seven years.
AbstractList Atypical lipomatous tumor (ALT)/well-differentiated liposarcoma, a relatively common malignant soft-tissue tumor, was first described in 1857 by Rudolf Virchow. It generally occurs in the four limbs and the retroperitoneum and rarely develops in the head and neck region. Here we report a case of an atypical lipomatous tumor in the left submandibular region. A 57-year-old man presented with a slowly enlarging mass in the lower left submandibular region. Initial examinations using computed tomography and magnetic resonance imaging revealed heterogeneous internal patterns; therefore, we performed an open biopsy. Histological examination of the specimen indicated lipoma. In February 2011, the patient underwent tumor excision under general anesthesia. The tumor adhered to the lingual nerve; we divided the tumor into parts to save the nerves and resected as much of the tumor as possible. Based on the histological examination of the resected specimens, an atypical lipomatous tumor was diagnosed. We re-operated in April to excise the remainder of the tumor. We excised it along with the lingual nerve via a mandibular swing approach because the tumor was in contact with the medial pterygoid muscle in the medial aspect of the mandible. No signs of recurrence were noted after seven years.
Author 森下, 佳学
長尾, 徹
鈴木, 慎太郎
後藤, 大輝
福村, 元洋
宮部, 悟
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References 4) Hirata M, Asano N, et al : Integrated exome and RNA sequencing of dedifferentiated liposarcoma. Nat Commun 10, 2019. Available at: https://www.nature.com/articles/s41467-019-13286-z Accessed June 9, 2020.
33) Priyal G, Shakil M, et al : Liposarcoma of the maxillary antrum: a case report. J Cancer Res Ther 11: 1023, 2015. Available at: http://www.cancerjournal.net/article.asp?issn=0973-1482;year=2015;volume=11;issue=4;spage=1023;epage=1023;aulast=Priyal Accessed June 9, 2020.
5) 中林成一郎， 鈴木秀明， 他：再発を繰り返した下咽頭脂肪肉腫例．耳鼻臨床 91: 699-704, 1998.
22) 峰晴昭仁， 川上理郎， 他：下咽頭原発の高分化型脂肪肉腫の1例．口腔咽頭科 22: 131-136, 2009.
25) Piperi E, Tosios KI, et al : Well-differentiated liposarcoma/atypical lipomatous tumor of the oral cavity: report of three cases and review of the literature. Head Neck Pathol 6: 354-363, 2012.
7) 日本整形外科学会診療ガイドライン委員会，軟部腫瘍診療ガイドライン策定委員会　編：軟部腫瘍診療ガイドライン2020. Available at: https://www.joa.or.jp/topics/2019/files/guideline_2020.pdf Accessed June 9, 2020.
10) 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 2002. 第3版，金原出版， 東京， 2002, 82-85 頁
40) 伊藤隆安， 中西啓介， 他： 脂肪系腫瘍に対するMRI， CTの有用性. 中部整災誌 35: 1677-1678, 1992.
15) Dubin MR and Chang EW : Liposarcoma of the tongue: case report and review of the literature. Head & Face Medicine 2, 2006. Available at: https://head-face-med.biomedcentral.com/track/pdf/10.1186/1746-160X-2-21 Accessed June 8, 2020.
42) Clay MR, Martinez AP, et al : MDM2 and CDK4 Immunohistochemistry: should it be used in problematic differentiated lipomatous tumors: a new perspective. Am J Surg Pathol 40: 1647-1652, 2016.
3) Fletcher CD, Bridge JA, et al : WHO Classification of Tumors of Soft Tissue and Bone. 4th Ed. IARC Press, Lyon, 2013, p33-43.
18) Titiz A, Yilmaz YF, et al : Atypical lipomatous tumor in the submental region. J Craniofac Surg 19: 1558-1560, 2008.
17) 大渕豊明， 宇高　毅， 他：頸部脂肪肉腫の1例．耳喉頭頸 78: 965-968, 2006.
23) Moritani N, Yamada T, et al : Atypical lipomatous tumor of the tongue: Report of a case. Acta Med. Okayama 64: 257-261, 2010.
21) DeWitt J, Heidelman J, et al : Atypical lipomatous tumors of the oral cavity: a report of 2 Cases. J Oral Maxillofac Surg 66: 366-369, 2008.
29) Kim YB, Leem DH, et al : Atypical lipomatous tumor/well-differentiated liposarcoma of the gingiva: a case report and review of literature. J Oral Maxillofac Surg 72: 431-439, 2014.
34) Nili F, Baghai F, et al : Well-differentiated liposarcoma of the floor of the mouth: report of a rare case and review of the literature. J Oral Maxillofac Pathol 20: 312-315, 2016.
26) Kaczmarczyk D, Jesionek-Kupnicka D, et al : Atypical lipomatous tumor of the cheek-a case report. Otolaryngol Pol 67: 218-221, 2013.
8) Binh MB, Sastre-Garau X, et al : MDM2 and CDK4 immunostainings are useful adjuncts in diagnosing well-differentiated and dedifferentiated liposarcoma subtypes: a comparative analysis of 559 soft tissue neoplasms with genetic data. Am J Surg Pathol 29: 1340-1347, 2005.
16) 道津　充， 畑地憲輔， 他：頸部多発性脂肪肉腫の1例．頭頸部外 16: 195-199, 2006.
41) Laco J, Mentzel T, et al : Atypical lipomatous tumors of the tongue: report of six cases. Virchows Arch 455: 383-388, 2009.
43) Weiss SW and Rao VK : Well-differentiated liposarcoma (atypical lipoma) of deep soft tissue of the extremities, retroperitoneum and miscellaneous sites: a follow-up study of 92 cases with analysis of the incidence of“ dedifferentiation”．Am J Surg Pathol 16: 1051-1058, 1992.
30) 平賀幸弘， 霜村真一， 他：耳下腺発生の高分化型脂肪肉腫症例．耳喉頭頸 86: 857-861, 2014.
36) 高橋　理， 山本雅史， 他：舌に発生し形態学的に異型脂肪腫様腫瘍と考えられた腫瘍の1例．日口外誌 65: 714-718, 2019.
44) Dey P : Fine needle aspiration cytology of well-differentiated liposarcoma. a report of two cases. Acta Cytol 44: 459-462, 2000.
28) Sotirović J, Vukomanović-Djurdjević B, et al : Recurrent lipomatous tumor of hypopharynx: case report and literature review. Acta Clin Croat 53: 365-368, 2014.
12) Nunes FD, Loducca SVL, et al : Case report: well-differentiated liposarcoma of the tongue. Oral Oncol 38: 117-119, 2002.
20) Paragis Sanchez T, Bannwart C, et al : Well-differentiated liposarcoma of the tongue. A case report. Minerva Stomatol 57: 383-387, 2008.
2) Golledge J, Fisher C, et al : Head and neck liposarcoma. Cancer 76: 1051-1058, 1995.
31) 正道隆介， 高橋　姿， 他：頭頸部領域に生じた高分化型脂肪肉腫の3例．頭頸部癌 40: 75-80, 2014.
39) 羽田孝司， 伊藤孝明， 他：後頸部に生じたatypical lipomatous tumor/well differentiated liposarcomaの1例. Skin Cancer 28: 24-28, 2013.
38) 堀　岳史， 安田剛敏， 他：意図的辺縁切除を行った高分化型脂肪肉腫の治療成績. 中部整災誌 56: 357-358, 2013.
24) Nouri H, Hassani R, et al : The well-differentiated liposarcoma of the hypopharynx. Eur Ann Otorhinolaryngol Head Neck Dis 128: 143-145, 2011.
6) Cheng J, Yu H, et al : Primary oral and maxillofacial liposarcoma: a clinicopathological and immunohistochemical study of eleven cases. Arch Med Sci 8: 316-232, 2012.
32) Kikuchi N, Nakashima T, et al : Well-differentiated liposarcoma arising in the parapharyngeal space: a case report and review of the literature. J Laryngo Otol 129: 86-90, 2015.
1) Enzinger FM, Weiss SW, et al : Soft Tissue Tumors. 3rd Ed. Mosby Co, St Louis, MO, 1995, p431-446.
37) Ohta K, Yoshimura H, et al : Oral liposarcoma in elderly: case report and literature analysis. Medicine Baltimore 99, 2020. Available at: https://www.ncbinlm.nih.gov/pmc/articles/PMC7015570/ Accessed June 8, 2020.
9) Gagari E, Kabani S, et al : Introral liposarcoma: case report and review of the literature. Oral Surg Oral Med Oral Pathol 89: 66-72, 2000.
11) 君塚　哲， 越後成志， 他：口腔に発生した脂肪肉腫の2例．日口外誌 54: 334-338, 2008.
35) Ioanidis KE, MacNeil SD, et al : An atypical lipomatous tumor mimicking a giant fibrovascular polyp of the hypopharynx. Medicine Baltimore 96, 2017. Available at: https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5671807/ Accessed June 9, 2020.
14) Allon I, Vered M, et al : Liposarcoma of the tongue: Clinico-pathologic correlations of a possible underdiagnosed entity. Oral Oncol 41: 657-665, 2005.
19) Chan WY, McHenry IDS, et al : Gingival liposarcoma: An unusual polyp. Br J Oral Maxillofac Surg 46: 150-151, 2008.
27) Pajaniappane A, Farzan J, et al : Well-differentiated liposarcoma of the epiglottis. J Laryngol Otol 128: 296-298, 2014.
13) Capodiferro S, Scully C, et al : Liposarcoma circumscriptum (lipoma-like) of the tongue: report of a case. Oral Dis 10: 398-400, 2004.
References_xml – reference: 42) Clay MR, Martinez AP, et al : MDM2 and CDK4 Immunohistochemistry: should it be used in problematic differentiated lipomatous tumors: a new perspective. Am J Surg Pathol 40: 1647-1652, 2016.
– reference: 6) Cheng J, Yu H, et al : Primary oral and maxillofacial liposarcoma: a clinicopathological and immunohistochemical study of eleven cases. Arch Med Sci 8: 316-232, 2012.
– reference: 27) Pajaniappane A, Farzan J, et al : Well-differentiated liposarcoma of the epiglottis. J Laryngol Otol 128: 296-298, 2014.
– reference: 33) Priyal G, Shakil M, et al : Liposarcoma of the maxillary antrum: a case report. J Cancer Res Ther 11: 1023, 2015. Available at: http://www.cancerjournal.net/article.asp?issn=0973-1482;year=2015;volume=11;issue=4;spage=1023;epage=1023;aulast=Priyal Accessed June 9, 2020.
– reference: 2) Golledge J, Fisher C, et al : Head and neck liposarcoma. Cancer 76: 1051-1058, 1995.
– reference: 14) Allon I, Vered M, et al : Liposarcoma of the tongue: Clinico-pathologic correlations of a possible underdiagnosed entity. Oral Oncol 41: 657-665, 2005.
– reference: 11) 君塚　哲， 越後成志， 他：口腔に発生した脂肪肉腫の2例．日口外誌 54: 334-338, 2008.
– reference: 15) Dubin MR and Chang EW : Liposarcoma of the tongue: case report and review of the literature. Head & Face Medicine 2, 2006. Available at: https://head-face-med.biomedcentral.com/track/pdf/10.1186/1746-160X-2-21 Accessed June 8, 2020.
– reference: 40) 伊藤隆安， 中西啓介， 他： 脂肪系腫瘍に対するMRI， CTの有用性. 中部整災誌 35: 1677-1678, 1992.
– reference: 43) Weiss SW and Rao VK : Well-differentiated liposarcoma (atypical lipoma) of deep soft tissue of the extremities, retroperitoneum and miscellaneous sites: a follow-up study of 92 cases with analysis of the incidence of“ dedifferentiation”．Am J Surg Pathol 16: 1051-1058, 1992.
– reference: 19) Chan WY, McHenry IDS, et al : Gingival liposarcoma: An unusual polyp. Br J Oral Maxillofac Surg 46: 150-151, 2008.
– reference: 21) DeWitt J, Heidelman J, et al : Atypical lipomatous tumors of the oral cavity: a report of 2 Cases. J Oral Maxillofac Surg 66: 366-369, 2008.
– reference: 34) Nili F, Baghai F, et al : Well-differentiated liposarcoma of the floor of the mouth: report of a rare case and review of the literature. J Oral Maxillofac Pathol 20: 312-315, 2016.
– reference: 44) Dey P : Fine needle aspiration cytology of well-differentiated liposarcoma. a report of two cases. Acta Cytol 44: 459-462, 2000.
– reference: 12) Nunes FD, Loducca SVL, et al : Case report: well-differentiated liposarcoma of the tongue. Oral Oncol 38: 117-119, 2002.
– reference: 29) Kim YB, Leem DH, et al : Atypical lipomatous tumor/well-differentiated liposarcoma of the gingiva: a case report and review of literature. J Oral Maxillofac Surg 72: 431-439, 2014.
– reference: 37) Ohta K, Yoshimura H, et al : Oral liposarcoma in elderly: case report and literature analysis. Medicine Baltimore 99, 2020. Available at: https://www.ncbinlm.nih.gov/pmc/articles/PMC7015570/ Accessed June 8, 2020.
– reference: 3) Fletcher CD, Bridge JA, et al : WHO Classification of Tumors of Soft Tissue and Bone. 4th Ed. IARC Press, Lyon, 2013, p33-43.
– reference: 5) 中林成一郎， 鈴木秀明， 他：再発を繰り返した下咽頭脂肪肉腫例．耳鼻臨床 91: 699-704, 1998.
– reference: 20) Paragis Sanchez T, Bannwart C, et al : Well-differentiated liposarcoma of the tongue. A case report. Minerva Stomatol 57: 383-387, 2008.
– reference: 4) Hirata M, Asano N, et al : Integrated exome and RNA sequencing of dedifferentiated liposarcoma. Nat Commun 10, 2019. Available at: https://www.nature.com/articles/s41467-019-13286-z Accessed June 9, 2020.
– reference: 41) Laco J, Mentzel T, et al : Atypical lipomatous tumors of the tongue: report of six cases. Virchows Arch 455: 383-388, 2009.
– reference: 13) Capodiferro S, Scully C, et al : Liposarcoma circumscriptum (lipoma-like) of the tongue: report of a case. Oral Dis 10: 398-400, 2004.
– reference: 16) 道津　充， 畑地憲輔， 他：頸部多発性脂肪肉腫の1例．頭頸部外 16: 195-199, 2006.
– reference: 28) Sotirović J, Vukomanović-Djurdjević B, et al : Recurrent lipomatous tumor of hypopharynx: case report and literature review. Acta Clin Croat 53: 365-368, 2014.
– reference: 35) Ioanidis KE, MacNeil SD, et al : An atypical lipomatous tumor mimicking a giant fibrovascular polyp of the hypopharynx. Medicine Baltimore 96, 2017. Available at: https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5671807/ Accessed June 9, 2020.
– reference: 18) Titiz A, Yilmaz YF, et al : Atypical lipomatous tumor in the submental region. J Craniofac Surg 19: 1558-1560, 2008.
– reference: 22) 峰晴昭仁， 川上理郎， 他：下咽頭原発の高分化型脂肪肉腫の1例．口腔咽頭科 22: 131-136, 2009.
– reference: 9) Gagari E, Kabani S, et al : Introral liposarcoma: case report and review of the literature. Oral Surg Oral Med Oral Pathol 89: 66-72, 2000.
– reference: 32) Kikuchi N, Nakashima T, et al : Well-differentiated liposarcoma arising in the parapharyngeal space: a case report and review of the literature. J Laryngo Otol 129: 86-90, 2015.
– reference: 36) 高橋　理， 山本雅史， 他：舌に発生し形態学的に異型脂肪腫様腫瘍と考えられた腫瘍の1例．日口外誌 65: 714-718, 2019.
– reference: 31) 正道隆介， 高橋　姿， 他：頭頸部領域に生じた高分化型脂肪肉腫の3例．頭頸部癌 40: 75-80, 2014.
– reference: 39) 羽田孝司， 伊藤孝明， 他：後頸部に生じたatypical lipomatous tumor/well differentiated liposarcomaの1例. Skin Cancer 28: 24-28, 2013.
– reference: 25) Piperi E, Tosios KI, et al : Well-differentiated liposarcoma/atypical lipomatous tumor of the oral cavity: report of three cases and review of the literature. Head Neck Pathol 6: 354-363, 2012.
– reference: 7) 日本整形外科学会診療ガイドライン委員会，軟部腫瘍診療ガイドライン策定委員会　編：軟部腫瘍診療ガイドライン2020. Available at: https://www.joa.or.jp/topics/2019/files/guideline_2020.pdf Accessed June 9, 2020.
– reference: 24) Nouri H, Hassani R, et al : The well-differentiated liposarcoma of the hypopharynx. Eur Ann Otorhinolaryngol Head Neck Dis 128: 143-145, 2011.
– reference: 26) Kaczmarczyk D, Jesionek-Kupnicka D, et al : Atypical lipomatous tumor of the cheek-a case report. Otolaryngol Pol 67: 218-221, 2013.
– reference: 17) 大渕豊明， 宇高　毅， 他：頸部脂肪肉腫の1例．耳喉頭頸 78: 965-968, 2006.
– reference: 23) Moritani N, Yamada T, et al : Atypical lipomatous tumor of the tongue: Report of a case. Acta Med. Okayama 64: 257-261, 2010.
– reference: 30) 平賀幸弘， 霜村真一， 他：耳下腺発生の高分化型脂肪肉腫症例．耳喉頭頸 86: 857-861, 2014.
– reference: 1) Enzinger FM, Weiss SW, et al : Soft Tissue Tumors. 3rd Ed. Mosby Co, St Louis, MO, 1995, p431-446.
– reference: 38) 堀　岳史， 安田剛敏， 他：意図的辺縁切除を行った高分化型脂肪肉腫の治療成績. 中部整災誌 56: 357-358, 2013.
– reference: 8) Binh MB, Sastre-Garau X, et al : MDM2 and CDK4 immunostainings are useful adjuncts in diagnosing well-differentiated and dedifferentiated liposarcoma subtypes: a comparative analysis of 559 soft tissue neoplasms with genetic data. Am J Surg Pathol 29: 1340-1347, 2005.
– reference: 10) 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 2002. 第3版，金原出版， 東京， 2002, 82-85 頁．
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Snippet Atypical lipomatous tumor (ALT)/well-differentiated liposarcoma, a relatively common malignant soft-tissue tumor, was first described in 1857 by Rudolf...
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異型脂肪腫様腫瘍
顎下部
Title MDM2遺伝子増幅を認めた顎下部異型脂肪腫様腫瘍の1例
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