線維性異形成症に続発した下顎低悪性骨中心性骨肉腫の1例
Sarcomatous transformation of fibrous dysplasia (FD) is an extremely rare event. We report a case of low-grade central osteosarcoma (LGCO), which arose secondarily from FD of the mandible. A 35-year-old woman, who had regularly been followed up for a 19-year period for a diagnosis of FD, presented w...
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| Published in | 日本口腔外科学会雑誌 Vol. 64; no. 7; pp. 423 - 428 |
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| Main Authors | , , , , , |
| Format | Journal Article |
| Language | Japanese |
| Published |
社団法人 日本口腔外科学会
20.07.2018
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| Subjects | |
| Online Access | Get full text |
| ISSN | 0021-5163 2186-1579 |
| DOI | 10.5794/jjoms.64.423 |
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| Abstract | Sarcomatous transformation of fibrous dysplasia (FD) is an extremely rare event. We report a case of low-grade central osteosarcoma (LGCO), which arose secondarily from FD of the mandible. A 35-year-old woman, who had regularly been followed up for a 19-year period for a diagnosis of FD, presented with a painful swelling in right mandibular body. On computed tomographic images, the right mental area was prominently expanded with the eroded cortex, and its inner part was entirely filled with an inhomogeneously enhanced lesion. Since a biopsy specimen taken from the anterior lesion was diagnosed to be LGCO, a segmental mandibulectomy and reconstruction with a fibula free flap was performed. There has been no sign of recurrence or metastasis as of 7 years after surgery. It is important to make a diagnosis and a treatment plan in patients with FD because malignant transformation of FD occurs rather infrequently. |
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| AbstractList | Sarcomatous transformation of fibrous dysplasia (FD) is an extremely rare event. We report a case of low-grade central osteosarcoma (LGCO), which arose secondarily from FD of the mandible. A 35-year-old woman, who had regularly been followed up for a 19-year period for a diagnosis of FD, presented with a painful swelling in right mandibular body. On computed tomographic images, the right mental area was prominently expanded with the eroded cortex, and its inner part was entirely filled with an inhomogeneously enhanced lesion. Since a biopsy specimen taken from the anterior lesion was diagnosed to be LGCO, a segmental mandibulectomy and reconstruction with a fibula free flap was performed. There has been no sign of recurrence or metastasis as of 7 years after surgery. It is important to make a diagnosis and a treatment plan in patients with FD because malignant transformation of FD occurs rather infrequently. |
| Author | 芳澤, 享子 西山, 秀昌 程, 珺 金丸, 祥平 新垣, 晋 小林, 正治 |
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| References | 4) Doganavsargil B, Argin M, et al : Fibrous: Secondary osteosarcoma arising in fibrous dysplasia, case report. Arch Orthop Trauma Surg 129: 439-44, 2009. 19) Mardinger O, Givol N, et al : Osteosarcoma of the jaw. The Chaim Sheba Medical Center experience. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 91: 445-451, 2001. 5) 石田 剛：Fibrous dysplasiaから発生した骨肉腫の1例．病理と臨 18: 114-115, 2000. 6) Ruggieri P, Sim FH, et al : Malignancies in fibrous dysplasia. Cancer 73: 1411-1424, 1994. 21) Sinha R, Chowdhury R, et al : Low-Grade Osteosarcoma of the Mandible. J Maxillofac Oral Surg 9: 186- 190, 2010. 13) Yoshida A, Ushiku T, et al : Immunohistochemical analysis of MDM2 and CDK4 distinguishes low-grade osteosarcoma from benign mimics. Mod Pathol 23: 1279-1288, 2010. 7) Yabut SM, Kenan S, et al : Malignant transformation of fibrous dysplasia. A case report and review of the literature. Clin Orthop Relat Res 228: 281-289, 1988. 9) Inwards C and Squire J : Low-grade central osteosarcoma. In Barnes L, Eveson JW, eds ; WHO Classification of Tumours, Pathology and Genetics of Bone Tumours. IARC Press, Lyon, 2005, p281-282. 8) Ebata K, Takeshi U, et al : Chondrosarcoma and osteosarcoma arising in polyostic fibrous dysplasia. J oral maxillofac surg 50: 761-764, 1992. 14) Pollandt K, Engels C, et al : Gsalpha gene mutations in monostotic fibrous dysplasia of bone and fibrous dysplasia-like low-grade central osteosarcoma. Virchows Arch 439: 170-175, 2001. 3) Heller AJ, DiNardo LJ, et al : Fibrous dysplasia, chondrosarcoma, and McCune-Albright syndrome. Am J Otolaryngol 22: 297-301, 2001. 11) Tabatabaei SH, Jahanshahi G, et al : Diagnostic challenges of low-grade central osteosarcoma of jaw: a literature review. J dent Shiraz Univ Med Sci 16: 62-67, 2015. 1) Albright F, Butter AM, et al : Syndrome characterized by posterities fibrosa disseminate, areas of pigmentation and endocrine dysfunction, with precocious puberty in females: report of five cases. N Engl J Med 216: 727-746, 1937. 2) Schwartz DT and Alpert M : The malignant transformation of fibrous dysplasia. Am J Med Sci 247: 1-20, 1964. 10) Cavalcanti MG, Ruprecht A, et al : Radiological findings in an unusual osteosarcoma in the maxilla. Dentomaxillofac Radiol 29: 180-184, 2000. 17) Andresen KJ, Sundaram M, et al : Imaging features of low-grade central osteosarcoma of the long bones and pelvis. Skeletal Radiol 33: 373-379, 2004. 15) Koyama T, Kobayashi T, et al : Radiation-induced undifferentiated high-grade pleomorphic sarcoma (malignant fibrous histiocytoma) of the mandible: report of a case arising in the background of long-standing osteomyelitis and review of the literature. Pathol Res Pract 210: 1123-1129, 2014. 12) Choong PF, Pritchard DJ, et al : Low grade central osteogenic sarcoma. A long-term followup of 20 patients. Clin Orthop Relat Res 322: 198-206, 1996. 18) Kurt AM, Unni KK, et al : Low-grade intraosseous osteosarcoma. Cancer 65: 1418-1428, 1990. 20) 日本小児血液がん学会：第3章 骨肉腫；小児がん診療ガイドライン．2016年版, 金原出版株式会社， 東京， 2016， 103-104 頁 16) Grivennikov SI, Greten FR, et al : Immunity, inflammation, and cancer. Clee 140: 883-899, 2010. |
| References_xml | – reference: 18) Kurt AM, Unni KK, et al : Low-grade intraosseous osteosarcoma. Cancer 65: 1418-1428, 1990. – reference: 1) Albright F, Butter AM, et al : Syndrome characterized by posterities fibrosa disseminate, areas of pigmentation and endocrine dysfunction, with precocious puberty in females: report of five cases. N Engl J Med 216: 727-746, 1937. – reference: 5) 石田 剛：Fibrous dysplasiaから発生した骨肉腫の1例．病理と臨 18: 114-115, 2000. – reference: 8) Ebata K, Takeshi U, et al : Chondrosarcoma and osteosarcoma arising in polyostic fibrous dysplasia. J oral maxillofac surg 50: 761-764, 1992. – reference: 2) Schwartz DT and Alpert M : The malignant transformation of fibrous dysplasia. Am J Med Sci 247: 1-20, 1964. – reference: 12) Choong PF, Pritchard DJ, et al : Low grade central osteogenic sarcoma. A long-term followup of 20 patients. Clin Orthop Relat Res 322: 198-206, 1996. – reference: 4) Doganavsargil B, Argin M, et al : Fibrous: Secondary osteosarcoma arising in fibrous dysplasia, case report. Arch Orthop Trauma Surg 129: 439-44, 2009. – reference: 17) Andresen KJ, Sundaram M, et al : Imaging features of low-grade central osteosarcoma of the long bones and pelvis. Skeletal Radiol 33: 373-379, 2004. – reference: 6) Ruggieri P, Sim FH, et al : Malignancies in fibrous dysplasia. Cancer 73: 1411-1424, 1994. – reference: 13) Yoshida A, Ushiku T, et al : Immunohistochemical analysis of MDM2 and CDK4 distinguishes low-grade osteosarcoma from benign mimics. Mod Pathol 23: 1279-1288, 2010. – reference: 19) Mardinger O, Givol N, et al : Osteosarcoma of the jaw. The Chaim Sheba Medical Center experience. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 91: 445-451, 2001. – reference: 3) Heller AJ, DiNardo LJ, et al : Fibrous dysplasia, chondrosarcoma, and McCune-Albright syndrome. Am J Otolaryngol 22: 297-301, 2001. – reference: 9) Inwards C and Squire J : Low-grade central osteosarcoma. In Barnes L, Eveson JW, eds ; WHO Classification of Tumours, Pathology and Genetics of Bone Tumours. IARC Press, Lyon, 2005, p281-282. – reference: 15) Koyama T, Kobayashi T, et al : Radiation-induced undifferentiated high-grade pleomorphic sarcoma (malignant fibrous histiocytoma) of the mandible: report of a case arising in the background of long-standing osteomyelitis and review of the literature. Pathol Res Pract 210: 1123-1129, 2014. – reference: 16) Grivennikov SI, Greten FR, et al : Immunity, inflammation, and cancer. Clee 140: 883-899, 2010. – reference: 7) Yabut SM, Kenan S, et al : Malignant transformation of fibrous dysplasia. A case report and review of the literature. Clin Orthop Relat Res 228: 281-289, 1988. – reference: 20) 日本小児血液がん学会：第3章 骨肉腫；小児がん診療ガイドライン．2016年版, 金原出版株式会社， 東京， 2016， 103-104 頁． – reference: 21) Sinha R, Chowdhury R, et al : Low-Grade Osteosarcoma of the Mandible. J Maxillofac Oral Surg 9: 186- 190, 2010. – reference: 11) Tabatabaei SH, Jahanshahi G, et al : Diagnostic challenges of low-grade central osteosarcoma of jaw: a literature review. J dent Shiraz Univ Med Sci 16: 62-67, 2015. – reference: 10) Cavalcanti MG, Ruprecht A, et al : Radiological findings in an unusual osteosarcoma in the maxilla. Dentomaxillofac Radiol 29: 180-184, 2000. – reference: 14) Pollandt K, Engels C, et al : Gsalpha gene mutations in monostotic fibrous dysplasia of bone and fibrous dysplasia-like low-grade central osteosarcoma. Virchows Arch 439: 170-175, 2001. |
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| Title | 線維性異形成症に続発した下顎低悪性骨中心性骨肉腫の1例 |
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