{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,29]],"date-time":"2026-07-29T02:48:27Z","timestamp":1785293307348,"version":"3.55.0"},"reference-count":18,"publisher":"Wiley","issue":"17","license":[{"start":{"date-parts":[[2025,9,9]],"date-time":"2025-09-09T00:00:00Z","timestamp":1757376000000},"content-version":"vor","delay-in-days":8,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100010735","name":"F\u00f6rderkreis Krebskranke Kinder e.V. Stuttgart","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100010735","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Cancer Medicine"],"published-print":{"date-parts":[[2025,9]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Purpose<\/jats:title>\n                    <jats:p>Teleangiectatic osteosarcoma is a histologic subtype of osteosarcoma that can mimic aneurysmal bone cysts and has so far been incompletely characterized.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Patients and Methods<\/jats:title>\n                    <jats:p>We used the database of the Cooperative Osteosarcoma Study Group COSS (patient\u2010registration 1980\u20132019) to better understand this rare histologic variant.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      223 eligible patients were identified, 164 having reference pathology (median age 15.9 (3.7\u201369.7) years; male 134, female 89; tumor sites limb 208 (201 metaphyses, 66 pathologic fractures), trunk 13, head &amp; neck 2; 26 with primary metastases). Known tumor\u2010predisposition syndromes were rare. Therapy included surgery in 215, radiotherapy in 10, and chemotherapy in all patients. Tumor response to preoperative treatment was good in 71% of 165 cases with available data. After a median follow\u2010up of 7.1 (0.2\u201332.1) years for all patients and 10.5 (0.2\u201332.1) years for 152 survivors, 5\u2010\/10\u2010year actuarial event\u2010free and overall survival expectancies were 61%\/57% and 73%\/66%, respectively. Five unrelated malignancies occurred during this period. The presence of primary metastases, pathologic fracture, poor response to neoadjuvant chemotherapy, and not obtaining a complete macroscopic remission were associated with inferior outcomes for both event\u2010free and overall survival (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&lt;\u20090.01).\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Discussion<\/jats:title>\n                    <jats:p>This large analysis proves teleangiectatic osteosarcoma to be a disease predominantly of the metaphyses of the young. While detected only rarely, the true incidence of genetic tumor predispositions would require prospective assessments. Behaving like other osteosarcoma subtypes in many other ways, this variant may show greater chemosensitivity and hence somewhat better outcomes than other subtypes.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/cam4.71211","type":"journal-article","created":{"date-parts":[[2025,9,9]],"date-time":"2025-09-09T09:55:27Z","timestamp":1757411727000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Teleangiectatic Osteosarcoma Treated by Surgery and Chemotherapy: A Report of 223 Affected Patients From the Cooperative Osteosarcoma Study Group (\n                    <scp>COSS<\/scp>\n                    )"],"prefix":"10.1002","volume":"14","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2144-3153","authenticated-orcid":false,"given":"Stefan S.","family":"Bielack","sequence":"first","affiliation":[{"name":"P\u00e4diatrie 5 (Onkologie, H\u00e4matologie, Immunologie), Stuttgart Cancer Center, Zentrum f\u00fcr Kinder\u2010, Jugend\u2010 und Frauenmedizin Klinikum Stuttgart \u2013 Olgahospital  Stuttgart Germany"},{"name":"Klinik f\u00fcr Kinder\u2010 und Jugendmedizin, P\u00e4diatrische H\u00e4matologie und Onkologie Universit\u00e4tsklinikum M\u00fcnster  M\u00fcnster Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4460-8830","authenticated-orcid":false,"given":"Vanessa","family":"Mettmann","sequence":"additional","affiliation":[{"name":"P\u00e4diatrie 5 (Onkologie, H\u00e4matologie, Immunologie), Stuttgart Cancer Center, Zentrum f\u00fcr Kinder\u2010, Jugend\u2010 und Frauenmedizin Klinikum Stuttgart \u2013 Olgahospital  Stuttgart Germany"},{"name":"Klinik f\u00fcr Kinder\u2010 und Jugendmedizin \u2013 H\u00e4matologie und Onkologie Universit\u00e4tsmedizin Mannheim  Mannheim Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2137-7507","authenticated-orcid":false,"given":"Daniel","family":"Baumhoer","sequence":"additional","affiliation":[{"name":"Knochentumor\u2010 Referenzzentrum, Institut f\u00fcr Medizinische Genetik und Pathologie Universit\u00e4tsspital Beider Basel, und Basel Research Centre for Child Health  Basel Switzerland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-3739-6093","authenticated-orcid":false,"given":"Andreas","family":"Beilken","sequence":"additional","affiliation":[{"name":"Kinder\u2010 und Jugendklinik, St\u00e4dtisches Klinikum Braunschweig gGmbH  Braunschweig Germany"},{"name":"Klinik f\u00fcr P\u00e4diatrische H\u00e4matologie und Onkologie Medizinische Hochschule Hannover  Hannover 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Universit\u00e4tsklinik f\u00fcr Kinder\u2010 und Jugendheilkunde der Medizinischen Universit\u00e4t Wien  Vienna Austria"},{"name":"St. Anna Children's Cancer Research Institute (CCRI)  Vienna Austria"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Matthias","family":"Kevric","sequence":"additional","affiliation":[{"name":"P\u00e4diatrie 5 (Onkologie, H\u00e4matologie, Immunologie), Stuttgart Cancer Center, Zentrum f\u00fcr Kinder\u2010, Jugend\u2010 und Frauenmedizin Klinikum Stuttgart \u2013 Olgahospital  Stuttgart Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8851-5014","authenticated-orcid":false,"given":"Thekla","family":"von Kalle","sequence":"additional","affiliation":[{"name":"Radiologisches Institut (Kinderradiologie), Zentrum f\u00fcr Kinder\u2010, Jugend\u2010 und Frauenmedizin, Klinikum Stuttgart \u2013 Olgahospital  Stuttgart Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1953-0848","authenticated-orcid":false,"given":"Andreas","family":"Kulozik","sequence":"additional","affiliation":[{"name":"Abteilung f\u00fcr P\u00e4diatrische Onkologie, H\u00e4matologie und Immunologie Universit\u00e4tsklinikum Heidelberg  Heidelberg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4523-1676","authenticated-orcid":false,"given":"Markus","family":"Metzler","sequence":"additional","affiliation":[{"name":"Kinder\u2010 und Jugendklinik \u2013 P\u00e4diatrische Onkologie und H\u00e4matologie Universit\u00e4tsklinikum Erlangen  Erlangen Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1584-1115","authenticated-orcid":false,"given":"Michaela","family":"Nathrath","sequence":"additional","affiliation":[{"name":"Klinik f\u00fcr P\u00e4diatrische H\u00e4matologie, Onkologie, Psychosomatik und Systemerkrankungen Klinikum Kassel  Kassel Germany"},{"name":"Kinderklinik der TU M\u00fcnchen\u2010Schwabing  Munich Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8672-5285","authenticated-orcid":false,"given":"Claudia","family":"Rossig","sequence":"additional","affiliation":[{"name":"Klinik f\u00fcr Kinder\u2010 und Jugendmedizin, P\u00e4diatrische H\u00e4matologie und Onkologie Universit\u00e4tsklinikum M\u00fcnster  M\u00fcnster Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Benjamin","family":"Sorg","sequence":"additional","affiliation":[{"name":"P\u00e4diatrie 5 (Onkologie, H\u00e4matologie, Immunologie), Stuttgart Cancer Center, Zentrum f\u00fcr Kinder\u2010, Jugend\u2010 und Frauenmedizin Klinikum Stuttgart \u2013 Olgahospital  Stuttgart 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