{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,17]],"date-time":"2026-08-17T15:57:37Z","timestamp":1786982257279,"version":"build-2736575974"},"reference-count":14,"publisher":"Springer Science and Business Media LLC","issue":"2","license":[{"start":{"date-parts":[[2020,9,21]],"date-time":"2020-09-21T00:00:00Z","timestamp":1600646400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2020,9,21]],"date-time":"2020-09-21T00:00:00Z","timestamp":1600646400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"name":"1","award":["Swiss National Science Foundation"],"award-info":[{"award-number":["Swiss National Science Foundation"]}]},{"name":"2","award":["the foundation of the Basel Bone Tumour Reference Centre"],"award-info":[{"award-number":["the foundation of the Basel Bone Tumour Reference Centre"]}]},{"name":"3","award":["the Gertrude von Meissner Stiftung"],"award-info":[{"award-number":["the Gertrude von Meissner Stiftung"]}]},{"name":"4","award":["the Stiftung f\u00fcr krebskranke Kinder, Regio Basiliensis."],"award-info":[{"award-number":["the Stiftung f\u00fcr krebskranke Kinder, Regio Basiliensis."]}]},{"DOI":"10.13039\/100008375","name":"University of Basel","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100008375","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Head and Neck Pathol"],"published-print":{"date-parts":[[2021,6]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>\n                    Cemento-osseous dysplasia (COD) is the most common benign fibro-osseous lesion of the jaws and generally considered non-neoplastic and self-limited. Here, we present a 30-year old female who noticed a bilateral swelling of her posterior mandible with irregular periapical mineralization and incomplete root resorption on panoramic radiographs. A biopsy revealed florid COD and no further treatment was initiated. 9 years later, she presented with a progressive expansion of her left posterior mandible after being treated for bilateral breast cancer 4 and 8 years before. CT scans showed expansile and densely mineralized lesions in all four quadrants with the left posterior mandible showing a focal penetration of the buccal cortical bone. Biopsies revealed an osteoblastic high-grade osteosarcoma in the left and a COD in the right mandible, notably with cellular atypia in the spindle cell component. The patient underwent segmental resection of the left mandible with clear margins and adjuvant chemotherapy. Subsequent genetic testing identified a heterozygous germline\n                    <jats:italic>TP53<\/jats:italic>\n                    mutation (p.V173G) which confirmed the clinically suspected Li\u2013Fraumeni syndrome (LFS). 3 years after the resection, the patient is free of disease and the other foci of COD remained stable in size on follow-up imaging analyses. Our case illustrates LFS-related osteosarcoma developing within florid COD. Given the rarity of this coincidence, a causative relation between the two lesions seems unlikely but in patients with tumor predisposition syndromes it might be advisable to closely monitor even benign lesions like COD.\n                  <\/jats:p>","DOI":"10.1007\/s12105-020-01223-2","type":"journal-article","created":{"date-parts":[[2020,9,21]],"date-time":"2020-09-21T16:02:25Z","timestamp":1600704145000},"page":"704-708","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Osteosarcoma of the Mandible in a Patient with Florid Cemento-Osseous Dysplasia and Li\u2013Fraumeni Syndrome: A Rare Coincidence"],"prefix":"10.1007","volume":"15","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4227-6051","authenticated-orcid":false,"given":"Simon","family":"Haefliger","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dorothee","family":"Harder","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michal","family":"Kovac","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karin","family":"Linkeschova","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Harald","family":"Eufinger","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daniel","family":"Baumhoer","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2020,9,21]]},"reference":[{"issue":"3","key":"1223_CR1","doi-asserted-by":"publisher","first-page":"141","DOI":"10.1259\/dmfr\/58488265","volume":"40","author":"NA Alsufyani","year":"2011","unstructured":"Alsufyani NA, Lam EW. Cemento-osseous dysplasia of the jaw bones: key radiographic features. Dentomaxillofac Radiol. 2011;40(3):141\u20136.","journal-title":"Dentomaxillofac Radiol"},{"issue":"1","key":"1223_CR2","doi-asserted-by":"publisher","first-page":"150","DOI":"10.4103\/0973-029X.110735","volume":"17","author":"BK Das","year":"2013","unstructured":"Das BK, Das SN, Gupta A, Nayak S. Florid cemento-osseous dysplasia. J Oral Maxillofac Pathol. 2013;17(1):150.","journal-title":"J Oral Maxillofac Pathol"},{"issue":"3","key":"1223_CR3","doi-asserted-by":"publisher","first-page":"466","DOI":"10.1007\/s12105-018-0992-5","volume":"13","author":"BL Nelson","year":"2019","unstructured":"Nelson BL, Phillips BJ. Benign fibro-osseous lesions of the head and neck. Head Neck Pathol. 2019;13(3):466\u201375.","journal-title":"Head Neck Pathol"},{"issue":"4","key":"1223_CR4","doi-asserted-by":"publisher","first-page":"432","DOI":"10.1007\/s12105-014-0590-0","volume":"8","author":"SK El-Mofty","year":"2014","unstructured":"El-Mofty SK. Fibro-osseous lesions of the craniofacial skeleton: an update. Head Neck Pathol. 2014;8(4):432\u201344.","journal-title":"Head Neck Pathol"},{"issue":"1","key":"1223_CR5","doi-asserted-by":"publisher","first-page":"159","DOI":"10.1007\/s00428-019-02726-2","volume":"476","author":"AHG Cleven","year":"2020","unstructured":"Cleven AHG, Schreuder WH, Groen E, Kroon HM, Baumhoer D. Molecular findings in maxillofacial bone tumours and its diagnostic value. Virchows Arch. 2020;476(1):159\u201374.","journal-title":"Virchows Arch"},{"key":"1223_CR6","first-page":"254","volume-title":"WHO classification of head and neck tumours","author":"SK El-Mofty","year":"2017","unstructured":"El-Mofty SK, Nelson B, Toyosawa S, Wright JM. Cemento-osseous dysplasia. In: El-Naggar AK, Chan JKC, Grandis JR, Takata T, Slootweg PJ, editors. WHO classification of head and neck tumours. Lyon: IARC Press; 2017. p. 254\u20135."},{"key":"1223_CR7","first-page":"251","volume-title":"WHO classification of head and neck tumours","author":"SK El-Mofty","year":"2017","unstructured":"El-Mofty SK, Nelson B, Toyosawa S. Ossifying fibroma. In: El-Naggar AK, Chan JKC, Grandis JR, Takata T, Slootweg PJ, editors. WHO classification of head and neck tumours. Lyon: IARC Press; 2017. p. 251\u20132."},{"key":"1223_CR8","first-page":"253","volume-title":"WHO classification of head and neck tumours","author":"SK El-Mofty","year":"2017","unstructured":"El-Mofty SK, Nelson B, Toyosawa S. Fibrous dysplasia. In: El-Naggar AK, Chan JKC, Grandis JR, Takata T, Slootweg PJ, editors. WHO classification of head and neck tumours. Lyon: IARC Press; 2017. p. 253\u20134."},{"issue":"2","key":"1223_CR9","doi-asserted-by":"publisher","first-page":"147","DOI":"10.1016\/j.oraloncology.2013.10.017","volume":"50","author":"D Baumhoer","year":"2014","unstructured":"Baumhoer D, Brunner P, Eppenberger-Castori S, Smida J, Nathrath M, Jundt G. Osteosarcomas of the jaws differ from their peripheral counterparts and require a distinct treatment approach. Experiences from the DOESAK Registry. Oral Oncol. 2014;50(2):147\u201353.","journal-title":"Oral Oncol"},{"key":"1223_CR10","first-page":"244","volume-title":"WHO classification of head and neck tumours","author":"D Baumhoer","year":"2017","unstructured":"Baumhoer D, Lopes M, Raubenheimer E. Osteosarcoma. In: El-Naggar AK, Chan JKC, Grandis JR, Takata T, Slootweg PJ, editors. WHO classification of head and neck tumours. Lyon: IARC Press; 2017. p. 244\u20136."},{"issue":"8","key":"1223_CR11","doi-asserted-by":"publisher","first-page":"785","DOI":"10.1016\/S1368-8375(02)00065-9","volume":"38","author":"YS Cheng","year":"2002","unstructured":"Cheng YS, Wright JM, Walstad WR, Finn MD. Osteosarcoma arising in Paget\u2019s disease of the mandible. Oral Oncol. 2002;38(8):785\u201392.","journal-title":"Oral Oncol"},{"issue":"4","key":"1223_CR12","doi-asserted-by":"publisher","first-page":"329","DOI":"10.1007\/s12105-010-0219-x","volume":"4","author":"MA Lopes","year":"2010","unstructured":"Lopes MA, Kim HS, Mariano FV, Correa MB, Rabelo NT, Vargas PA. Clinico-pathologic conference: case 1. High-grade osteosarcoma (OS) and florid cemento-osseous dysplasia (FCOD). Head Neck Pathol. 2010;4(4):329\u201333.","journal-title":"Head Neck Pathol"},{"issue":"3","key":"1223_CR13","doi-asserted-by":"publisher","first-page":"296","DOI":"10.1016\/S1079-2104(03)00416-5","volume":"96","author":"R Melrose","year":"2003","unstructured":"Melrose R, Handlers J. Osteosarcoma ex florid osseous dysplasia: report of a case. Oral Surg Oral Med Oral Pathol Oral Radiol Endodontol. 2003;96(3):296.","journal-title":"Oral Surg Oral Med Oral Pathol Oral Radiol Endodontol"},{"issue":"1","key":"1223_CR14","doi-asserted-by":"publisher","first-page":"69","DOI":"10.1016\/S1079-2104(99)70195-2","volume":"88","author":"LC Schneider","year":"1999","unstructured":"Schneider LC, Dolinsky HB, Grodjesk JE, Mesa ML, Doyle JL. Malignant spindle cell tumor arising in the mandible of a patient with florid osseous dysplasia. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1999;88(1):69\u201373.","journal-title":"Oral Surg Oral Med Oral Pathol Oral Radiol Endod"}],"container-title":["Head and Neck Pathology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s12105-020-01223-2.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1007\/s12105-020-01223-2\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s12105-020-01223-2.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,9,20]],"date-time":"2021-09-20T20:27:15Z","timestamp":1632169635000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1007\/s12105-020-01223-2"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,9,21]]},"references-count":14,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2021,6]]}},"alternative-id":["1223"],"URL":"https:\/\/doi.org\/10.1007\/s12105-020-01223-2","relation":{},"ISSN":["1936-0568"],"issn-type":[{"value":"1936-0568","type":"electronic"}],"subject":[],"published":{"date-parts":[[2020,9,21]]},"assertion":[{"value":"7 August 2020","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"2 September 2020","order":2,"name":"revised","label":"Revised","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"3 September 2020","order":3,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"21 September 2020","order":4,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Compliance with Ethical Standards"}},{"value":"The authors declare that they have no conflict of interest.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of interest"}},{"value":"Written consent was obtained from the patient.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Informed Consent"}}]}}