{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,11]],"date-time":"2026-07-11T02:25:19Z","timestamp":1783736719869,"version":"3.55.0"},"reference-count":23,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2020,7,16]],"date-time":"2020-07-16T00:00:00Z","timestamp":1594857600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2020,7,16]],"date-time":"2020-07-16T00:00:00Z","timestamp":1594857600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"name":"Beijing Talents Fund","award":["2017000021469G239"],"award-info":[{"award-number":["2017000021469G239"]}]},{"name":"Academic New Star Program of Beijing Jishuitan Hospital","award":["No XKXX201610"],"award-info":[{"award-number":["No XKXX201610"]}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["J Orthop Surg Res"],"published-print":{"date-parts":[[2020,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n<jats:title>Background<\/jats:title>\n<jats:p>The clinical and molecular characteristics of osteofibrous dysplasia (OFD)-like adamantinoma (AD) differ from those of classic AD. Most reports about OFD-like AD are case reports or small case series. More cases from different centers are still warranted.<\/jats:p>\n<\/jats:sec><jats:sec>\n<jats:title>Methods<\/jats:title>\n<jats:p>The orthopedic oncology database of our institution was searched to identify patients with AD. The cases of OFD-like and classic AD of the long bones were retrospectively analyzed. Between December 1999 and August 2016, 23 patients were treated for AD, comprising seven with OFD-like AD and 16 with classic AD. The outcomes were compared between AD subtypes.<\/jats:p>\n<\/jats:sec><jats:sec>\n<jats:title>Results<\/jats:title>\n<jats:p>In the OFD-like AD group, four lesions were treated with extensive curettage, while three were treated with wide resection. The median follow-up duration in the OFD-like AD group was 66\u2009months (range 43\u2013131\u2009months). At the end of follow-up, there was only one case of local recurrence (LR) in the OFD-like AD group, giving a LR rate of 14.3% (1\/7). No distant metastasis or progression to classic AD was detected in the OFD-like AD group. In the classic AD group, the treatments were below-the-knee amputation in one patient with extensive tibial and fibular lesions, curettage with a bone graft in one patient who was diagnosed with OFD based on a core needle biopsy, hemi-cortical excision and reconstruction in two patients, and segmental resection and reconstruction in 12 patients. At the end of follow-up, there were three cases of LR in the classic AD group, giving a LR rate of 18.8% (3\/16); two patients developed lung metastasis after LR and died of the disease at 88 and 126\u2009months after the first surgery in our hospital, respectively. The classic AD group had a metastatic rate of 12.5% (2\/16), a final limb salvage rate of 75%, and estimated 5- and 10-year survival rates of 88.9% and 77.1%, respectively.<\/jats:p>\n<\/jats:sec><jats:sec>\n<jats:title>Conclusions<\/jats:title>\n<jats:p>OFD-like AD has a better outcome than classic AD. For OFD-like AD, extensive curettage is suggested if the tumor extent allows. For classic AD, aggressive resection with wide margins is essential to achieve local control. A long-term follow-up is necessary due to the possibility of late complications.<\/jats:p>\n<\/jats:sec>","DOI":"10.1186\/s13018-020-01769-5","type":"journal-article","created":{"date-parts":[[2020,7,16]],"date-time":"2020-07-16T12:02:47Z","timestamp":1594900967000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":13,"title":["Outcome of osteofibrous dysplasia-like versus classic adamantinoma of long bones: a single-institution experience"],"prefix":"10.1186","volume":"15","author":[{"given":"Zhiping","family":"Deng","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lihua","family":"Gong","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Qing","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lin","family":"Hao","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yi","family":"Ding","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiaohui","family":"Niu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2020,7,16]]},"reference":[{"issue":"4","key":"1769_CR1","doi-asserted-by":"publisher","first-page":"704","DOI":"10.2106\/00004623-195436040-00002","volume":"36-a","author":"PL Baker","year":"1954","unstructured":"Baker PL, Dockerty MB, Coventry MB. Adamantinoma (so-called) of the long bones; review of the literature and report of three new cases. J Bone Joint Surg Am. 1954;36-a(4):704\u201320.","journal-title":"J Bone Joint Surg Am"},{"issue":"1","key":"1769_CR2","doi-asserted-by":"publisher","first-page":"6823","DOI":"10.4081\/rt.2017.6823","volume":"9","author":"S Yoshida","year":"2017","unstructured":"Yoshida S, Murakami T, Suzuki K, Itou S, Watanuki M, Hosaka M, et al. Adamantinoma arising in the distal end of the fibula. Rare tumors. 2017;9(1):6823.","journal-title":"Rare tumors."},{"issue":"2","key":"1769_CR3","doi-asserted-by":"publisher","first-page":"152","DOI":"10.1007\/s00776-003-0753-4","volume":"9","author":"S Ii","year":"2004","unstructured":"Ii S, Tsuchiya H, Takazawa K, Minato H, Tomita K. Adamantinoma of the proximal femur: a case report. J Orthop Sci. 2004;9(2):152\u20136.","journal-title":"J Orthop Sci"},{"key":"1769_CR4","doi-asserted-by":"publisher","first-page":"256","DOI":"10.1097\/00003086-200303000-00034","volume":"408","author":"GM Sherman","year":"2003","unstructured":"Sherman GM, Damron TA, Yang Y. CD99 positive adamantinoma of the ulna with ipsilateral discrete osteofibrous dysplasia. Clinical orthopaedics and related research. 2003;408:256\u201361.","journal-title":"Clinical orthopaedics and related research."},{"issue":"8","key":"1769_CR5","doi-asserted-by":"crossref","first-page":"1121","DOI":"10.3928\/0147-7447-19890801-13","volume":"12","author":"RP Clarke","year":"1989","unstructured":"Clarke RP, Leonard JR, von Kuster L, Wesseler TA. Adamantinoma of the humerus with early metastases and death. A case report with autopsy findings. Orthopedics. 1989;12(8):1121\u20135.","journal-title":"Orthopedics."},{"key":"1769_CR6","doi-asserted-by":"crossref","unstructured":"Chandrasekar CR, Mohammed R, Rafalla AA, Grimer RJ. Adamantinoma of the calcaneum--a case report. Foot (Edinburgh, Scotland). 2009;19(1):58\u201361.","DOI":"10.1016\/j.foot.2008.09.004"},{"key":"1769_CR7","first-page":"234","volume":"309","author":"DS Springfield","year":"1994","unstructured":"Springfield DS, Rosenberg AE, Mankin HJ, Mindell ER. Relationship between osteofibrous dysplasia and adamantinoma. Clin Orthop Relat Res. 1994;309:234\u201344.","journal-title":"Clin Orthop Relat Res"},{"issue":"6","key":"1769_CR8","doi-asserted-by":"publisher","first-page":"815","DOI":"10.1302\/0301-620X.91B6.21577","volume":"91","author":"BG Spiegelberg","year":"2009","unstructured":"Spiegelberg BG, Sewell MD, Coltman T, Blunn GW, Flanagan AM, Cannon SR, et al. Below-knee amputation through a joint-sparing proximal tibial replacement for recurrent tumour. J Bone Joint Surg Br. 2009;91(6):815\u20139.","journal-title":"J Bone Joint Surg Br"},{"issue":"3","key":"1769_CR9","doi-asserted-by":"publisher","first-page":"409","DOI":"10.1302\/0301-620X.99B3.38050","volume":"99-B","author":"DW Scholfield","year":"2017","unstructured":"Scholfield DW, Sadozai Z, Ghali C, Sumathi V, Douis H, Gaston L, et al. Does osteofibrous dysplasia progress to adamantinoma and how should they be treated? Bone Joint J. 2017;99-B(3):409\u201316.","journal-title":"Bone Joint J"},{"issue":"7","key":"1769_CR10","doi-asserted-by":"publisher","first-page":"965","DOI":"10.1097\/PAS.0000000000001251","volume":"43","author":"NM Ali","year":"2019","unstructured":"Ali NM, Niada S, Morris MR, Brini AT, Huen D, Sumathi V, et al. Comprehensive molecular characterization of adamantinoma and OFD-like adamantinoma bone tumors. Am J Surg Pathol. 2019;43(7):965\u201374.","journal-title":"Am J Surg Pathol"},{"issue":"5","key":"1769_CR11","first-page":"1","volume":"11","author":"M Ondhia","year":"2018","unstructured":"Ondhia M, Garg N, Sumathi V, Harave S. Paediatric osteofibrous dysplasia-like adamantinoma with classical radiological findings. BMJ case reports. 2018;11(5):1\u20133.","journal-title":"BMJ case reports."},{"issue":"2","key":"1769_CR12","first-page":"210","volume":"49","author":"H Buldu","year":"2015","unstructured":"Buldu H, Centel T, Kirimlioglu H, Dirik Y. Osteofibrous dysplasia-like adamantinoma in a 3-month-old male infant: a case report. Acta Orthop Traumatol Turc. 2015;49(2):210\u20132.","journal-title":"Acta Orthop Traumatol Turc"},{"issue":"10","key":"1769_CR13","first-page":"E411","volume":"44","author":"A Ratra","year":"2015","unstructured":"Ratra A, Wooldridge A, Brindley G. Osteofibrous dysplasia-like adamantinoma of the tibia in a 15-year-old girl. Am J Orthop. 2015;44(10):E411\u20133.","journal-title":"Am J Orthop"},{"issue":"10","key":"1769_CR14","doi-asserted-by":"publisher","first-page":"1482","DOI":"10.2106\/00004623-199410000-00008","volume":"76","author":"HM Hazelbag","year":"1994","unstructured":"Hazelbag HM, Taminiau AH, Fleuren GJ, Hogendoorn PC. Adamantinoma of the long bones. A clinicopathological study of thirty-two patients with emphasis on histological subtype, precursor lesion, and biological behavior. J Bone Joint Surg Am. 1994;76(10):1482\u201399.","journal-title":"J Bone Joint Surg Am"},{"key":"1769_CR15","first-page":"215","volume":"204","author":"NF Moon","year":"1986","unstructured":"Moon NF, Mori H. Adamantinoma of the appendicular skeleton--updated. Clin Orthop Relat Res. 1986;204:215\u201337.","journal-title":"Clin Orthop Relat Res"},{"issue":"2","key":"1769_CR16","doi-asserted-by":"publisher","first-page":"193","DOI":"10.1177\/1066896918790388","volume":"27","author":"B Rekhi","year":"2019","unstructured":"Rekhi B, Sahay A, Puri A. Clinicopathologic features of two rare cases of dedifferentiated adamantinomas, including diagnostic implications. Int J Surg Pathol. 2019;27(2):193\u2013202.","journal-title":"Int J Surg Pathol"},{"issue":"9","key":"1769_CR17","doi-asserted-by":"publisher","first-page":"1388","DOI":"10.1097\/PAS.0b013e3181ecfe6a","volume":"34","author":"FM Izquierdo","year":"2010","unstructured":"Izquierdo FM, Ramos LR, Sanchez-Herraez S, Hernandez T, de Alava E, Hazelbag HM. Dedifferentiated classic adamantinoma of the tibia: a report of a case with eventual complete revertant mesenchymal phenotype. Am J Surg Pathol. 2010;34(9):1388\u201392.","journal-title":"Am J Surg Pathol"},{"issue":"7","key":"1769_CR18","doi-asserted-by":"publisher","first-page":"770","DOI":"10.1016\/j.otsr.2011.05.005","volume":"97","author":"H Nouri","year":"2011","unstructured":"Nouri H, Jaafoura H, Bouaziz M, Ouertatani M, Abid L, Meherzi MH, et al. Dedifferentiated adamantinoma associated with fibrous dysplasia. Orthop Traumatol Surg Res. 2011;97(7):770\u20135.","journal-title":"Orthop Traumatol Surg Res"},{"issue":"1","key":"1769_CR19","doi-asserted-by":"publisher","first-page":"53","DOI":"10.1620\/tjem.209.53","volume":"209","author":"M Hatori","year":"2006","unstructured":"Hatori M, Watanabe M, Hosaka M, Sasano H, Narita M, Kokubun S. A classic adamantinoma arising from osteofibrous dysplasia-like adamantinoma in the lower leg: a case report and review of the literature. Tohoku J Exp Med. 2006;209(1):53\u20139.","journal-title":"Tohoku J Exp Med"},{"issue":"8","key":"1769_CR20","doi-asserted-by":"publisher","first-page":"809","DOI":"10.1016\/S0046-8177(98)90449-X","volume":"29","author":"G Kuruvilla","year":"1998","unstructured":"Kuruvilla G, Steiner GC. Osteofibrous dysplasia-like adamantinoma of bone: a report of five cases with immunohistochemical and ultrastructural studies. Human pathology. 1998;29(8):809\u201314.","journal-title":"Human pathology."},{"issue":"7","key":"1769_CR21","doi-asserted-by":"publisher","first-page":"1150","DOI":"10.1002\/jso.25269","volume":"118","author":"MT Houdek","year":"2018","unstructured":"Houdek MT, Sherman CE, Inwards CY, Wenger DE, Rose PS, Sim FH. Adamantinoma of bone: long-term follow-up of 46 consecutive patients. J Surg Oncol. 2018;118(7):1150\u20134.","journal-title":"J Surg Oncol"},{"issue":"7","key":"1769_CR22","doi-asserted-by":"publisher","first-page":"925","DOI":"10.1016\/j.otsr.2016.08.010","volume":"102","author":"SE Puchner","year":"2016","unstructured":"Puchner SE, Varga R, Hobusch GM, Kasparek M, Panotopoulos J, Lang S, et al. Long-term outcome following treatment of adamantinoma and osteofibrous dysplasia of long bones. Orthop Traumatol Surg Res. 2016;102(7):925\u201332.","journal-title":"Orthop Traumatol Surg Res"},{"issue":"8","key":"1769_CR23","doi-asserted-by":"publisher","first-page":"1122","DOI":"10.2106\/00004623-200008000-00009","volume":"82","author":"AA Qureshi","year":"2000","unstructured":"Qureshi AA, Shott S, Mallin BA, Gitelis S. Current trends in the management of adamantinoma of long bones. An international study. J Bone Joint Surg Am. 2000;82(8):1122\u201331.","journal-title":"J Bone Joint Surg Am"}],"container-title":["Journal of Orthopaedic Surgery and Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s13018-020-01769-5.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s13018-020-01769-5\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s13018-020-01769-5.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,7,15]],"date-time":"2021-07-15T23:27:27Z","timestamp":1626391647000},"score":1,"resource":{"primary":{"URL":"https:\/\/josr-online.biomedcentral.com\/articles\/10.1186\/s13018-020-01769-5"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,7,16]]},"references-count":23,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2020,12]]}},"alternative-id":["1769"],"URL":"https:\/\/doi.org\/10.1186\/s13018-020-01769-5","relation":{},"ISSN":["1749-799X"],"issn-type":[{"value":"1749-799X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2020,7,16]]},"assertion":[{"value":"19 April 2020","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"30 June 2020","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"16 July 2020","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"The study protocol was approved by the ethics committee of Beijing Jishuitan Hospital.","order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Not applicable.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no competing interests.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"268"}}