{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T14:45:12Z","timestamp":1791816312358,"version":"4.3.4"},"reference-count":39,"publisher":"Wiley","license":[{"start":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T00:00:00Z","timestamp":1791763200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"},{"start":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T00:00:00Z","timestamp":1791763200000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pediatric Blood &amp;amp; Cancer"],"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Outcomes in relapsed or refractory pediatric and AYA bone sarcomas remain poor, and the impact of early referral to specialized sarcoma centers on survival is unclear. Early centralization of care may improve local and systemic treatment quality, but evidence in this population is limited.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>This secondary analysis of a prospective single\u2010center regorafenib trial in children and adolescents\/young adults with relapsed or refractory bone sarcomas compared outcomes between patients managed at a specialized high\u2010volume sarcoma center from initial diagnosis (\u201cinternal\u201d group) and those referred only at relapse (\u201cexternal\u201d group). Baseline characteristics, prior treatments, and trial exposure were assessed. Overall survival (OS) and event\u2010free survival (EFS) were estimated from trial enrollment using Kaplan\u2013Meier methods and compared between groups.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Thirty patients were included (18 internal, 12 external). Baseline clinical features, including histologic subtype, metastatic status at diagnosis, and time from diagnosis to first relapse\/progression, were broadly similar between groups. EFS from trial enrollment did not differ significantly between groups. However, 24\u2010month OS was longer in patients managed at the specialized center from diagnosis (77% vs. 42%), suggesting a persistent influence of earlier phases of care despite equal access to protocol\u2010based relapse therapy at study entry.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Early centralization of care at a specialized pediatric and AYA sarcoma center may confer a long\u2010term survival advantage in relapsed or refractory bone sarcomas, even when investigational relapse therapy is standardized. These findings support timely referral to expert centers and may inform the organization of care and referral pathways for pediatric and AYA patients with bone sarcomas.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/1545-5017.70756","type":"journal-article","created":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T13:51:18Z","timestamp":1791813078000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Impact of Initial Treatment Setting on Survival in Relapsed Pediatric and AYA Bone Sarcomas: A Secondary Analysis of a Prospective Clinical Trial"],"prefix":"10.1002","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8629-1165","authenticated-orcid":false,"given":"Justyna","family":"Antoniuk\u2010Majchrzak","sequence":"first","affiliation":[{"name":"Department of Surgical Oncology for Children and Youth Institute of Mother and Child  Warsaw Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katarzyna","family":"Bilska","sequence":"additional","affiliation":[{"name":"Department of Surgical Oncology for Children and Youth Institute of Mother and Child  Warsaw Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tomasz","family":"Kozi\u0144ski","sequence":"additional","affiliation":[{"name":"Department of Surgical Oncology for Children and Youth Institute of Mother and Child  Warsaw Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jadwiga","family":"W\u0119c\u0142awek\u2010Tompol","sequence":"additional","affiliation":[{"name":"Department and Clinic of Pediatric Oncology Hematology and Bone Marrow Transplantation Wroclaw Medical University  Warsaw Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dorota","family":"Sega\u2010Pondel","sequence":"additional","affiliation":[{"name":"Department and Clinic of Pediatric Oncology Hematology and Bone Marrow Transplantation Wroclaw Medical University  Warsaw Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rados\u0142aw","family":"Chaber","sequence":"additional","affiliation":[{"name":"Department of Pediatric Oncohematology Rzeszow University  Rzeszow Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wanda","family":"Badowska","sequence":"additional","affiliation":[{"name":"Department of Pediatric Oncology and Hematology Department of Clinical Pediatrics University of Warmia &amp; Mazury Provincial Specialist Children's Hospital  Olsztyn Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katarzyna","family":"Ros\u0142an","sequence":"additional","affiliation":[{"name":"Department of Pediatric Oncology and Hematology Medical University of Bialystok Children's Clinical Hospital of L. Zamenhof  Bia\u0142ystok Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hanna","family":"Kose\u0142a\u2010Paterczyk","sequence":"additional","affiliation":[{"name":"Department of Soft Tissue\/Bone Sarcoma and Melanoma Maria Sklodowska\u2010Curie National Research Institute of Oncology  Warsaw Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2612-7934","authenticated-orcid":false,"given":"Anna","family":"Raciborska","sequence":"additional","affiliation":[{"name":"Department of Surgical Oncology for Children and Youth Institute of Mother and Child  Warsaw Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,10,12]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1177\/2333794x211042238"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejca.2006.05.015"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.5603\/njo.105973"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.6061\/clinics\/2019\/e858"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.mayocp.2012.01.015"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1002\/ijc.24320"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1097\/MOP.0b013e32832b1111"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.3390\/cancers13061202"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1302\/1863\u20102548.15.210085"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.5435\/JAAOSGlobal\u2010D\u201024\u201000281"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1136\/archdischild\u20102024\u2010327879"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.4103\/0971\u20105851.203513"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00264\u2010006\u20100192\u2010x"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejcped.2023.100029"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.28355"},{"key":"e_1_2_9_17_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.28352"},{"key":"e_1_2_9_18_1","doi-asserted-by":"publisher","DOI":"10.5312\/wjo.v7.i9.527"},{"key":"e_1_2_9_19_1","doi-asserted-by":"publisher","DOI":"10.4103\/0019\u20105413.69305"},{"key":"e_1_2_9_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.suronc.2016.03.006"},{"key":"e_1_2_9_21_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.27763"},{"key":"e_1_2_9_22_1","doi-asserted-by":"publisher","DOI":"10.5435\/JAAOSGlobal\u2010D\u201024\u201000130"},{"key":"e_1_2_9_23_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.27444"},{"key":"e_1_2_9_24_1","doi-asserted-by":"publisher","DOI":"10.1186\/1477\u20107819\u201010\u2010191"},{"key":"e_1_2_9_25_1","doi-asserted-by":"publisher","DOI":"10.1302\/1863\u20102548.15.210107"},{"key":"e_1_2_9_26_1","doi-asserted-by":"publisher","DOI":"10.3238\/arztebl.m2023.0079"},{"key":"e_1_2_9_27_1","doi-asserted-by":"publisher","DOI":"10.1089\/jayao.2021.0110"},{"key":"e_1_2_9_28_1","doi-asserted-by":"publisher","DOI":"10.3390\/cancers16213616"},{"key":"e_1_2_9_29_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12885\u2010023\u201010556\u20104"},{"key":"e_1_2_9_30_1","doi-asserted-by":"publisher","DOI":"10.12865\/CHSJ.50.03.03"},{"key":"e_1_2_9_31_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12913\u2010020\u201005532\u20108"},{"key":"e_1_2_9_32_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.28998"},{"key":"e_1_2_9_33_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0158406"},{"key":"e_1_2_9_34_1","doi-asserted-by":"publisher","DOI":"10.2147\/JMDH.S49805"},{"key":"e_1_2_9_35_1","doi-asserted-by":"publisher","DOI":"10.1089\/jayao.2018.0136"},{"key":"e_1_2_9_36_1","doi-asserted-by":"publisher","DOI":"10.1186\/1472\u20106963\u201012\u201082"},{"key":"e_1_2_9_37_1","doi-asserted-by":"publisher","DOI":"10.1245\/s10434\u2010019\u201007421\u20109"},{"key":"e_1_2_9_38_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.critrevonc.2016.12.002"},{"key":"e_1_2_9_39_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.esmoop.2024.103734"},{"key":"e_1_2_9_40_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00423\u2010023\u201003002\u20103"}],"container-title":["Pediatric Blood &amp; 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