{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,13]],"date-time":"2026-03-13T16:23:54Z","timestamp":1773419034329,"version":"3.50.1"},"reference-count":40,"publisher":"Oxford University Press (OUP)","issue":"3","license":[{"start":{"date-parts":[[2025,11,30]],"date-time":"2025-11-30T00:00:00Z","timestamp":1764460800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/pages\/standard-publication-reuse-rights"}],"funder":[{"DOI":"10.13039\/501100000646","name":"JSPS","doi-asserted-by":"publisher","award":["JP23K14978"],"award-info":[{"award-number":["JP23K14978"]}],"id":[{"id":"10.13039\/501100000646","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,3,13]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Pazopanib is used to treat relapsed and refractory sarcomas. Pazopanib\u2019s role in pediatric, adolescent, and young adult populations remains unestablished.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>To assess pazopanib\u2019s utility, we analyzed retrospectively collected data from pediatric (0\u201314\u00a0years) and adolescent and young adult (15\u201339\u00a0years) patients diagnosed with relapsed or refractory sarcomas who received pazopanib.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>We assessed data from 21 patients (10 pediatric, 11 adolescent, and young adult). Their diagnoses included osteosarcoma (n\u00a0=\u00a011), rhabdomyosarcoma (n\u00a0=\u00a04), alveolar soft part sarcoma (n\u00a0=\u00a05), and leiomyosarcoma (n\u00a0=\u00a01). Thirteen (62%) patients presented with metastatic disease at the initial diagnosis. Patients had received a median of three prior chemotherapy regimens (range: 0\u20136). The median duration of pazopanib treatment was 3.5\u00a0months (range: 1\u201312) for pediatric patients and 4\u00a0months (range: 1\u201383) for adolescents and young adults. Nine patients (five adolescents and young adults) discontinued pazopanib owing to disease progression, and two discontinued owing to adverse events (pneumothorax). We observed seven cases of stable disease (four adolescents and young adults) and 12 of progressive disease (six adolescents and young adults) after ~3\u00a0months. The median survival following pazopanib initiation was 7.8, 4.8, and 12.4\u00a0months for overall, pediatric, and adolescent and young adult patients, respectively.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>In a small cohort of children and adolescent and young adult patients with heavily pretreated relapsed or refractory sarcoma, pazopanib may be a feasible option. Further research on optimal therapeutic timing and the target population for pazopanib\u2019s indication is required.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1093\/jjco\/hyaf191","type":"journal-article","created":{"date-parts":[[2025,11,16]],"date-time":"2025-11-16T12:55:22Z","timestamp":1763297722000},"page":"309-316","source":"Crossref","is-referenced-by-count":0,"title":["Pazopanib therapy for children, adolescents, and young adults with relapsed and refractory sarcomas"],"prefix":"10.1093","volume":"56","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1675-7038","authenticated-orcid":false,"given":"Kosuke","family":"Tamefusa","sequence":"first","affiliation":[{"name":"Department of Pediatrics, Okayama University Hospital , 2-5-1 Shikata-cho, Kita-ku, Okayama 700-8558,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hisashi","family":"Ishida","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Okayama University Hospital , 2-5-1 Shikata-cho, Kita-ku, Okayama 700-8558,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tomohiro","family":"Fujiwara","sequence":"additional","affiliation":[{"name":"Department of Orthopedic Surgery, Okayama University Hospital , 2-5-1 Shikata-cho, Kita-ku, Okayama 700-8558 ,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Go","family":"Makimoto","sequence":"additional","affiliation":[{"name":"Center for Clinical Oncology, Okayama University Hospital , 2-5-1 Shikata-cho, Kita-ku, Okayama 700-8558 ,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Motoharu","family":"Ochi","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Okayama University Hospital , 2-5-1 Shikata-cho, Kita-ku, Okayama 700-8558,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takahiro","family":"Shiwaku","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Okayama University Hospital , 2-5-1 Shikata-cho, Kita-ku, Okayama 700-8558,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kaori","family":"Fujiwara","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Okayama University Hospital , 2-5-1 Shikata-cho, Kita-ku, Okayama 700-8558,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yasuhisa","family":"Tatebe","sequence":"additional","affiliation":[{"name":"Department of Pharmacy, Okayama University Hospital , 2-5-1 Shikata-cho, Kita-ku, Okayama 700-8558 ,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kana","family":"Washio","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Okayama University Hospital , 2-5-1 Shikata-cho, Kita-ku, Okayama 700-8558,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Masahiro","family":"Tabata","sequence":"additional","affiliation":[{"name":"Center for Clinical Oncology, Okayama University Hospital , 2-5-1 Shikata-cho, Kita-ku, Okayama 700-8558 ,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Toshifumi","family":"Ozaki","sequence":"additional","affiliation":[{"name":"Department of Orthopedic Surgery, Okayama University Hospital , 2-5-1 Shikata-cho, Kita-ku, Okayama 700-8558 ,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hirokazu","family":"Tsukahara","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Okayama University Hospital , 2-5-1 Shikata-cho, Kita-ku, Okayama 700-8558,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2025,11,30]]},"reference":[{"key":"2026031310020713800_ref1","doi-asserted-by":"publisher","first-page":"675","DOI":"10.1200\/JCO.23.01275","article-title":"Soft tissue sarcomas in adolescents and young adults","volume":"42","author":"Weiss","year":"2024","journal-title":"J Clin Oncol"},{"key":"2026031310020713800_ref2","doi-asserted-by":"publisher","first-page":"e28279","DOI":"10.1002\/pbc.28279","article-title":"Non-rhabdomyosarcoma soft-tissue sarcoma","volume":"68","author":"Milgrom","year":"2021","journal-title":"Pediatr Blood Cancer"},{"key":"2026031310020713800_ref3","doi-asserted-by":"publisher","first-page":"10","DOI":"10.1016\/j.ejca.2022.03.028","article-title":"The treatment approach to pediatric non-rhabdomyosarcoma soft tissue sarcomas: A critical review from the INternational soft tissue SaRcoma ConsorTium","volume":"169","author":"Ferrari","year":"2022","journal-title":"Eur J Cancer"},{"key":"2026031310020713800_ref4","doi-asserted-by":"publisher","first-page":"254","DOI":"10.1001\/jamaoncol.2018.4490","article-title":"Diagnosis, prognosis, and treatment of alveolar soft-part sarcoma: A review: A review","volume":"5","author":"Paoluzzi","year":"2019","journal-title":"JAMA Oncol"},{"key":"2026031310020713800_ref5","doi-asserted-by":"publisher","first-page":"3029","DOI":"10.1200\/JCO.2014.59.4895","article-title":"Osteosarcoma: Current treatment and a collaborative pathway to success","volume":"33","author":"Isakoff","year":"2015","journal-title":"J Clin Oncol"},{"key":"2026031310020713800_ref6","doi-asserted-by":"publisher","first-page":"188","DOI":"10.1200\/JCO.2017.75.1743","article-title":"Osteosarcoma, chondrosarcoma, and chordoma","volume":"36","author":"Whelan","year":"2018","journal-title":"J Clin Oncol"},{"key":"2026031310020713800_ref7","doi-asserted-by":"publisher","first-page":"2066","DOI":"10.1056\/NEJMra2103423","article-title":"New horizons in the treatment of osteosarcoma","volume":"385","author":"Meltzer","year":"2021","journal-title":"N Engl J Med"},{"key":"2026031310020713800_ref8","doi-asserted-by":"publisher","first-page":"7","DOI":"10.1007\/s10147-021-02064-x","article-title":"Cancer in adolescents and young adults in Japan: Epidemiology and cancer strategy","volume":"27","author":"Nakata","year":"2022","journal-title":"Int J Clin Oncol"},{"key":"2026031310020713800_ref9","doi-asserted-by":"publisher","first-page":"559","DOI":"10.1200\/JCO.2005.04.063","article-title":"Osteosarcoma relapse after combined modality therapy: An analysis of unselected patients in the cooperative osteosarcoma study group (COSS)","volume":"23","author":"Kempf-Bielack","year":"2005","journal-title":"J Clin Oncol"},{"key":"2026031310020713800_ref10","doi-asserted-by":"publisher","first-page":"3031","DOI":"10.1200\/JCO.2015.65.5381","article-title":"Outcome of patients with recurrent osteosarcoma enrolled in seven phase II trials through children\u2019s cancer group, pediatric oncology group, and Children\u2019s oncology group: Learning from the past to move forward","volume":"34","author":"Lagmay","year":"2016","journal-title":"J Clin Oncol"},{"key":"2026031310020713800_ref11","doi-asserted-by":"publisher","first-page":"503","DOI":"10.1093\/jjco\/hyy053","article-title":"Drug review: Pazopanib","volume":"48","author":"Miyamoto","year":"2018","journal-title":"Jpn J Clin Oncol"},{"key":"2026031310020713800_ref12","doi-asserted-by":"publisher","first-page":"1879","DOI":"10.1016\/S0140-6736(12)60651-5","article-title":"Pazopanib for metastatic soft-tissue sarcoma (PALETTE): A randomised, double-blind, placebo-controlled phase 3 trial","volume":"379","author":"Graaf","year":"2012","journal-title":"Lancet"},{"key":"2026031310020713800_ref13","doi-asserted-by":"publisher","first-page":"1110","DOI":"10.1016\/S1470-2045(20)30325-9","article-title":"Pathological response in children and adults with large unresected intermediate-grade or high-grade soft tissue sarcoma receiving preoperative chemoradiotherapy with or without pazopanib (ARST1321): A multicentre, randomised, open-label, phase 2 trial","volume":"21","author":"Weiss","year":"2020","journal-title":"Lancet Oncol"},{"key":"2026031310020713800_ref14","doi-asserted-by":"publisher","first-page":"248","DOI":"10.1093\/jjco\/hyv184","article-title":"A randomized, double-blind, placebo-controlled, phase III study of pazopanib in patients with soft tissue sarcoma: Results from the Japanese subgroup","volume":"46","author":"Kawai","year":"2016","journal-title":"Jpn J Clin Oncol"},{"key":"2026031310020713800_ref15","doi-asserted-by":"publisher","first-page":"3034","DOI":"10.1200\/JCO.2012.47.0914","article-title":"Phase I pharmacokinetic and pharmacodynamic study of pazopanib in children with soft tissue sarcoma and other refractory solid tumors: A children\u2019s oncology group phase I consortium report","volume":"31","author":"Glade Bender","year":"2013","journal-title":"J Clin Oncol"},{"key":"2026031310020713800_ref16","doi-asserted-by":"publisher","first-page":"1228","DOI":"10.3389\/fonc.2020.01228","article-title":"A chart review on the feasibility and safety of the vincristine irinotecan pazopanib (VIPaz) association in children and adolescents with resistant or relapsed sarcomas","volume":"10","author":"Russo","year":"2020","journal-title":"Front Oncol"},{"key":"2026031310020713800_ref17","doi-asserted-by":"publisher","first-page":"937","DOI":"10.1111\/ped.13307","article-title":"Pazopanib for second recurrence of osteosarcoma in pediatric patients","volume":"59","author":"Umeda","year":"2017","journal-title":"Pediatr Int"},{"key":"2026031310020713800_ref18","doi-asserted-by":"publisher","first-page":"228","DOI":"10.1016\/j.ejca.2008.10.026","article-title":"New response evaluation criteria in solid tumours: Revised RECIST guideline (version 1.1)","volume":"45","author":"Eisenhauer","year":"2009","journal-title":"Eur J Cancer"},{"key":"2026031310020713800_ref19","doi-asserted-by":"publisher","first-page":"452","DOI":"10.1038\/bmt.2012.244","article-title":"Investigation of the freely available easy-to-use software \u201cEZR\u201d for medical statistics","volume":"48","author":"Kanda","year":"2013","journal-title":"Bone Marrow Transplant"},{"key":"2026031310020713800_ref20","doi-asserted-by":"publisher","first-page":"397","DOI":"10.1007\/s00776-009-1347-6","article-title":"Multiinstitutional phase II study of neoadjuvant chemotherapy for osteosarcoma (NECO study) in Japan: NECO-93J and NECO-95J","volume":"14","author":"Iwamoto","year":"2009","journal-title":"J Orthop Sci"},{"key":"2026031310020713800_ref21","doi-asserted-by":"publisher","first-page":"1396","DOI":"10.1016\/S1470-2045(16)30214-5","article-title":"Comparison of MAPIE versus MAP in patients with a poor response to preoperative chemotherapy for newly diagnosed high-grade osteosarcoma (EURAMOS-1): An open-label, international, randomised controlled trial","volume":"17","author":"Marina","year":"2016","journal-title":"Lancet Oncol"},{"key":"2026031310020713800_ref22","doi-asserted-by":"publisher","first-page":"5","DOI":"10.1002\/pbc.24118","article-title":"Rhabdomyosarcoma: Review of the Children\u2019s oncology group (COG) soft-tissue sarcoma committee experience and rationale for current COG studies: COG rhabdomyosarcoma experience","volume":"59","author":"Malempati","year":"2012","journal-title":"Pediatr Blood Cancer"},{"key":"2026031310020713800_ref23","doi-asserted-by":"publisher","first-page":"117","DOI":"10.1200\/JCO.2015.63.4048","article-title":"Intensive multiagent therapy, including dose-compressed cycles of Ifosfamide\/etoposide and vincristine\/doxorubicin\/cyclophosphamide, irinotecan, and radiation, in patients with high-risk rhabdomyosarcoma: A report from the Children\u2019s oncology group","volume":"34","author":"Weigel","year":"2016","journal-title":"J Clin Oncol"},{"key":"2026031310020713800_ref24","doi-asserted-by":"publisher","first-page":"e28323","DOI":"10.1002\/pbc.28323","article-title":"A case of alveolar rhabdomyosarcoma showing concurrent responsive bone marrow lesions and refractory pancreatic lesions to pazopanib monotherapy","volume":"67","author":"Kanamitsu","year":"2020","journal-title":"Pediatr Blood Cancer"},{"key":"2026031310020713800_ref25","doi-asserted-by":"publisher","first-page":"14","DOI":"10.1186\/2045-3329-4-14","article-title":"Pneumothorax as adverse event in patients with lung metastases of soft tissue sarcoma treated with pazopanib: A single reference Centre case series","volume":"4","author":"Verschoor","year":"2014","journal-title":"Clin Sarcoma Res"},{"key":"2026031310020713800_ref26","doi-asserted-by":"publisher","first-page":"750","DOI":"10.1186\/s12885-016-2786-z","article-title":"Risk factors for pneumothorax in advanced and\/or metastatic soft tissue sarcoma patients during pazopanib treatment: A single-institute analysis","volume":"16","author":"Nakano","year":"2016","journal-title":"BMC Cancer"},{"key":"2026031310020713800_ref27","doi-asserted-by":"publisher","first-page":"e0254866","DOI":"10.1371\/journal.pone.0254866","article-title":"Different patterns of pneumothorax in patients with soft tissue tumors treated with pazopanib: A case series analysis","volume":"16","author":"Aiba","year":"2021","journal-title":"PloS One"},{"key":"2026031310020713800_ref28","doi-asserted-by":"publisher","first-page":"891","DOI":"10.1186\/s12885-022-09968-5","article-title":"Alveolar soft part sarcoma: Progress toward improvement in survival? A population-based study","volume":"22","author":"Fujiwara","year":"2022","journal-title":"BMC Cancer"},{"key":"2026031310020713800_ref29","doi-asserted-by":"publisher","first-page":"20","DOI":"10.1634\/theoncologist.2018-0464","article-title":"A phase II trial of pazopanib in patients with metastatic alveolar soft part sarcoma","volume":"24","author":"Kim","year":"2019","journal-title":"Oncologist"},{"key":"2026031310020713800_ref30","doi-asserted-by":"publisher","first-page":"911","DOI":"10.1056\/NEJMoa2303383","article-title":"Atezolizumab for advanced alveolar soft part sarcoma","volume":"389","author":"Chen","year":"2023","journal-title":"N Engl J Med"},{"key":"2026031310020713800_ref31","doi-asserted-by":"publisher","first-page":"4842","DOI":"10.1200\/JCO.23.00045","article-title":"Outcomes after preoperative chemoradiation with or without pazopanib in non-rhabdomyosarcoma soft tissue sarcoma: A report from children\u2019s oncology group and NRG oncology","volume":"41","author":"Weiss","year":"2023","journal-title":"J Clin Oncol"},{"key":"2026031310020713800_ref32","doi-asserted-by":"publisher","first-page":"3080","DOI":"10.1111\/1759-7714.14669","article-title":"Pazopanib for treating rhabdomyosarcoma in adult patients with poor performance status: A case report","volume":"13","author":"Nishii","year":"2022","journal-title":"Thorac Cancer"},{"key":"2026031310020713800_ref33","doi-asserted-by":"publisher","first-page":"508","DOI":"10.1093\/annonc\/mdr151","article-title":"A phase II trial of sorafenib in relapsed and unresectable high-grade osteosarcoma after failure of standard multimodal therapy: An Italian sarcoma group study","volume":"23","author":"Grignani","year":"2012","journal-title":"Ann Oncol"},{"key":"2026031310020713800_ref34","doi-asserted-by":"publisher","first-page":"1732","DOI":"10.1016\/S1470-2045(16)30507-1","article-title":"Safety and efficacy of regorafenib in patients with advanced soft tissue sarcoma (REGOSARC): A randomised, double-blind, placebo-controlled, phase 2 trial","volume":"17","author":"Mir","year":"2016","journal-title":"Lancet Oncol"},{"key":"2026031310020713800_ref35","doi-asserted-by":"publisher","first-page":"120","DOI":"10.1016\/S1470-2045(18)30742-3","article-title":"Efficacy and safety of regorafenib in adult patients with metastatic osteosarcoma: A non-comparative, randomised, double-blind, placebo-controlled, phase 2 study","volume":"20","author":"Duffaud","year":"2019","journal-title":"Lancet Oncol"},{"key":"2026031310020713800_ref36","doi-asserted-by":"publisher","first-page":"142","DOI":"10.1016\/j.ejca.2021.05.023","article-title":"Phase 1 dose-escalation and pharmacokinetic study of regorafenib in paediatric patients with recurrent or refractory solid malignancies","volume":"153","author":"Geoerger","year":"2021","journal-title":"Eur J Cancer"},{"key":"2026031310020713800_ref37","doi-asserted-by":"publisher","first-page":"1940","DOI":"10.1038\/s41416-023-02413-9","article-title":"Regorafenib in patients with advanced Ewing sarcoma: Results of a non-comparative, randomised, double-blind, placebo-controlled, multicentre phase II study","volume":"129","author":"Duffaud","year":"2023","journal-title":"Br J Cancer"},{"key":"2026031310020713800_ref38","doi-asserted-by":"publisher","first-page":"446","DOI":"10.1016\/S1470-2045(19)30825-3","article-title":"Cabozantinib in patients with advanced Ewing sarcoma or osteosarcoma (CABONE): A multicentre, single-arm, phase 2 trial","volume":"21","author":"Italiano","year":"2020","journal-title":"Lancet Oncol"},{"key":"2026031310020713800_ref39","doi-asserted-by":"publisher","first-page":"100250","DOI":"10.1016\/j.esmoop.2021.100250","article-title":"Phase I\/II study of single-agent lenvatinib in children and adolescents with refractory or relapsed solid malignancies and young adults with osteosarcoma (ITCC-050)\u2606","volume":"6","author":"Gaspar","year":"2021","journal-title":"ESMO Open"},{"key":"2026031310020713800_ref40","doi-asserted-by":"publisher","first-page":"1645","DOI":"10.1001\/jamaoncol.2024.4381","article-title":"Lenvatinib plus ifosfamide and etoposide in children and young adults with relapsed osteosarcoma: A phase 2 randomized clinical trial","volume":"10","author":"Gaspar","year":"2024","journal-title":"JAMA Oncol"}],"container-title":["Japanese Journal of Clinical Oncology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/jjco\/article-pdf\/56\/3\/309\/65661222\/hyaf191.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/jjco\/article-pdf\/56\/3\/309\/65661222\/hyaf191.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,3,13]],"date-time":"2026-03-13T14:02:15Z","timestamp":1773410535000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/jjco\/article\/56\/3\/309\/8361808"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,11,30]]},"references-count":40,"journal-issue":{"issue":"3","published-online":{"date-parts":[[2025,11,30]]},"published-print":{"date-parts":[[2026,3,13]]}},"URL":"https:\/\/doi.org\/10.1093\/jjco\/hyaf191","relation":{},"ISSN":["1465-3621"],"issn-type":[{"value":"1465-3621","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2026,3]]},"published":{"date-parts":[[2025,11,30]]}}}