{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,15]],"date-time":"2026-02-15T08:03:41Z","timestamp":1771142621327,"version":"3.50.1"},"reference-count":22,"publisher":"Wiley","issue":"9","license":[{"start":{"date-parts":[[2025,5,9]],"date-time":"2025-05-09T00:00:00Z","timestamp":1746748800000},"content-version":"vor","delay-in-days":8,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2025,5,1]],"date-time":"2025-05-01T00:00:00Z","timestamp":1746057600000},"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":["Cancer Medicine"],"published-print":{"date-parts":[[2025,5]]},"abstract":"ABSTRACT<\/jats:title>\n \n Background<\/jats:title>\n Patients with relapsed or refractory soft tissue and bone sarcomas have dismal outcomes. Multi\u2010targeted kinase inhibitors (mTKI) have proven to be potent agents in several malignancies, both as primer therapy and as a salvage option. Our aim was to evaluate the clinical outcomes of mTKI treatment in a heterogeneous group of pediatric sarcoma patients retrospectively.<\/jats:p>\n <\/jats:sec>\n \n Procedures<\/jats:title>\n A total of 18 patients were treated with sorafenib, regorafenib, or pazopanib; 13 of them had osteosarcoma (OSC), 3 had synovial sarcoma (SySa), and 1\u20131 patient had chondrosarcoma and rhabdomyosarcoma. Indication for mTKI treatment was primarily progressive, inoperable, relapsed, or chemotherapy\u2010resistant disease after completion of first\u2010 and second\u2010line chemotherapy.<\/jats:p>\n <\/jats:sec>\n \n Results<\/jats:title>\n \n At the time of the beginning of mTKI treatment, the median age was 16.5\u2009years, and the median time to progression from initiation of mTKI was 4\u2009months. The overall response rate was 16%. We conducted a comparison of the survival outcomes of OSC patients receiving mTKIs against a retrospective, non\u2010randomized control group. Overall survival was evaluated from the time of progression or relapse after second\u2010line treatment to the time of death. The log\u2010rank test revealed a significant difference in the survival distribution between patients receiving mTKIs and those who did not (chi2(1)\u2009=\u20098.13\n p<\/jats:italic>\n \u2009=\u20090.004). We observed benefits from mTKI treatment in 3 SySa patients, with pazopanib demonstrating effectiveness and no progression observed thus far.\n <\/jats:p>\n <\/jats:sec>\n \n Conclusions<\/jats:title>\n Our findings suggest that mTKIs are well\u2010tolerated and can serve as a therapeutic option for refractory bone sarcomas as palliative treatment, aiming to slow disease progression and uphold a good quality of life.<\/jats:p>\n <\/jats:sec>","DOI":"10.1002\/cam4.70951","type":"journal-article","created":{"date-parts":[[2025,5,9]],"date-time":"2025-05-09T07:20:03Z","timestamp":1746775203000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Multi\u2010Targeted Kinase Inhibitor Therapy in Pediatric Bone and Soft Tissue Sarcoma Patients\u2014A Single Centre Experience"],"prefix":"10.1002","volume":"14","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2021-823X","authenticated-orcid":false,"given":"Anna","family":"Moh\u00e1s","sequence":"first","affiliation":[{"name":"Pediatric Centre, T\u0171zolt\u00f3 Street Department Semmelweis University Budapest Hungary"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kl\u00e1ra","family":"Horv\u00e1th","sequence":"additional","affiliation":[{"name":"Pediatric Centre, T\u0171zolt\u00f3 Street Department Semmelweis University Budapest Hungary"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Zsuzsanna","family":"Jakab","sequence":"additional","affiliation":[{"name":"National Childhood Cancer Registry (NCCR), Hungarian Pediatric Oncology Network (HuPON). Department of Paediatrics Semmelweis University Budapest Hungary"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Monika","family":"Cs\u00f3ka","sequence":"additional","affiliation":[{"name":"Pediatric Centre, T\u0171zolt\u00f3 Street Department Semmelweis University Budapest Hungary"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2025,5,9]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejca.2018.11.027"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41572-018-0051-2"},{"key":"e_1_2_11_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.23252"},{"key":"e_1_2_11_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jbo.2017.07.001"},{"key":"e_1_2_11_6_1","doi-asserted-by":"publisher","DOI":"10.1093\/annonc\/mdr151"},{"key":"e_1_2_11_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1470-2045(14)71136-2"},{"key":"e_1_2_11_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(12)61900-X"},{"key":"e_1_2_11_9_1","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.18.02374"},{"key":"e_1_2_11_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(12)60651-5"},{"key":"e_1_2_11_11_1","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.2002.20.3.776"},{"key":"e_1_2_11_12_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.29691"},{"key":"e_1_2_11_13_1","doi-asserted-by":"publisher","DOI":"10.1093\/jnci\/djn"},{"key":"e_1_2_11_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.phrs.2016.12.004"},{"key":"e_1_2_11_15_1","doi-asserted-by":"publisher","DOI":"10.3389\/fcell.2020.00613"},{"key":"e_1_2_11_16_1","doi-asserted-by":"publisher","DOI":"10.3389\/fonc.2020.01642"},{"key":"e_1_2_11_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1470-2045(20)30325-9"},{"key":"e_1_2_11_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejca.2022.06.008"},{"key":"e_1_2_11_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1470-2045(18)30742-3"},{"key":"e_1_2_11_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1470-2045(19)30825-3"},{"key":"e_1_2_11_21_1","doi-asserted-by":"publisher","DOI":"10.1093\/annonc\/mdt586"},{"key":"e_1_2_11_22_1","doi-asserted-by":"publisher","DOI":"10.2217\/fon-2022-0348"},{"key":"e_1_2_11_23_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140\u20106736(16)32453\u20109"}],"container-title":["Cancer Medicine"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/cam4.70951","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/cam4.70951","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/cam4.70951","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,2,15]],"date-time":"2026-02-15T07:15:46Z","timestamp":1771139746000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/cam4.70951"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,5]]},"references-count":22,"journal-issue":{"issue":"9","published-print":{"date-parts":[[2025,5]]}},"alternative-id":["10.1002\/cam4.70951"],"URL":"https:\/\/doi.org\/10.1002\/cam4.70951","archive":["Portico"],"relation":{},"ISSN":["2045-7634","2045-7634"],"issn-type":[{"value":"2045-7634","type":"print"},{"value":"2045-7634","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,5]]},"assertion":[{"value":"2024-08-25","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-04-29","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-05-09","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e70951"}}