{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,8]],"date-time":"2026-03-08T19:09:58Z","timestamp":1772996998591,"version":"3.50.1"},"reference-count":26,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2026,1,29]],"date-time":"2026-01-29T00:00:00Z","timestamp":1769644800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,1,29]],"date-time":"2026-01-29T00:00:00Z","timestamp":1769644800000},"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 Reports"],"published-print":{"date-parts":[[2026,2]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>We present the case of 72\u2013year\u2010old male with metastatic de\u2010differentiated chondrosarcoma (\u201cDDCS\u201d). DDCS is a rare soft tissue cancer that carries a dismal prognosis in the metastatic stage, and is resistant to both traditional chemotherapy and radiotherapy. There is a distinct lack of proven systemic therapies.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case<\/jats:title>\n                    <jats:p>This case report is distinguished in that our patient had a significant clinical response to combination therapy with an immune checkpoint inhibitor (Pembrolizumab) and a multi\u2010targeted tyrosine kinase inhibitor (Pazopanib). Our patient presented with bony pain after suffering a pathological fracture of the left humerus after grabbing a fence. He did not report prior constitutional symptoms or bony pain. On examination he was clinically in atrial fibrillation and reported reduced exercise tolerance with a New York Heart association grading of 2. There were no other pertinent clinical findings. FDG PET\u2010CT scan revealed a 10cmx5cm destructive intra\u2010osseous lesion of the proximal humerus, markedly FDG\u2010avid, without evidence of metastatic disease. He underwent immediate surgical resection, followed by adjuvant radiotherapy to the left humerus. Tumour histology revealed a high\u2010grade DDCS. Genetic sequencing revealed alterations in IDH2, PTCH1 and TERT promoter. Restaging FDG PET scan 3 months after diagnosis revealed lung metastases. The patient was commenced on Vismodegib with best disease response of progressive disease. Eight months after diagnosis he was commenced on combination therapy of Pazopanib and Pembrolizumab, with significant reduction in size of lung metastases. He sustained a progression free period of 6 months on this regime. Treatment course was complicated primarily by hepatotoxicity, which resolved with dose reduction of Pazopanib. Eleven months after diagnosis, FDG PET\u2010CT revealed relapse and significant progression of metastatic disease and systemic therapy was ceased. The patient passed away 14 months after diagnosis.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>This case report is a valuable example of promising emerging systemic therapies for advanced DDCS, where the present standard of care lacks a repertoire of effective therapies.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/cnr2.70426","type":"journal-article","created":{"date-parts":[[2026,1,29]],"date-time":"2026-01-29T12:31:17Z","timestamp":1769689877000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["A Case Report of Promising Response to Combination Therapy With an Immune Checkpoint Inhibitor (Pembrolizumab) and Multi\u2010Targeted Tyrosine Kinase Inhibitor (Pazopanib) in Metastatic De\u2010Differentiated Chondrosarcoma"],"prefix":"10.1002","volume":"9","author":[{"ORCID":"https:\/\/orcid.org\/0009-0000-7182-585X","authenticated-orcid":false,"given":"Matthew","family":"Youssef","sequence":"first","affiliation":[{"name":"Royal Prince Alfred Hospital  Sydney New South Wales Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2653-1657","authenticated-orcid":false,"given":"Peter","family":"Grimison","sequence":"additional","affiliation":[{"name":"Royal Prince Alfred Hospital  Sydney New South Wales Australia"},{"name":"Chris O'Brien Lifehouse  Sydney New South Wales Australia"},{"name":"University of Sydney  Sydney New South Wales Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2026,1,29]]},"reference":[{"key":"e_1_2_13_2_1","doi-asserted-by":"publisher","DOI":"10.1002\/jor.24463"},{"key":"e_1_2_13_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/cncr.33618"},{"key":"e_1_2_13_4_1","doi-asserted-by":"publisher","DOI":"10.1007\/s40487-024-00317-z"},{"key":"e_1_2_13_5_1","doi-asserted-by":"publisher","DOI":"10.6004\/jnccn.2025.0017"},{"key":"e_1_2_13_6_1","doi-asserted-by":"publisher","DOI":"10.3389\/fonc.2022.991724"},{"key":"e_1_2_13_7_1","doi-asserted-by":"publisher","DOI":"10.1177\/17588359231199877"},{"key":"e_1_2_13_8_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamaoncol.2020.3689"},{"key":"e_1_2_13_9_1","doi-asserted-by":"publisher","DOI":"10.3390\/cancers15092617"},{"key":"e_1_2_13_10_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41379-022-01008-8"},{"key":"e_1_2_13_11_1","doi-asserted-by":"publisher","DOI":"10.1051\/sicotj\/2025011"},{"key":"e_1_2_13_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jbo.2022.100456"},{"key":"e_1_2_13_13_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13018-024-05098-9"},{"key":"e_1_2_13_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.annonc.2021.08.1995"},{"key":"e_1_2_13_15_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11999-013-3360-5"},{"key":"e_1_2_13_16_1","doi-asserted-by":"publisher","DOI":"10.1302\/0301-620X.82B1.0820055"},{"issue":"4","key":"e_1_2_13_17_1","first-page":"35448","article-title":"Dedifferentiated Chondrosarcoma: A Case Series and Review of the Literature","volume":"14","author":"Gusho C. A.","year":"2022","journal-title":"Orthopedic Reviews (Pavia)"},{"key":"e_1_2_13_18_1","doi-asserted-by":"publisher","DOI":"10.5301\/tj.5000696"},{"key":"e_1_2_13_19_1","doi-asserted-by":"publisher","DOI":"10.1093\/annonc\/mdt374"},{"key":"e_1_2_13_20_1","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.19.02492"},{"key":"e_1_2_13_21_1","doi-asserted-by":"publisher","DOI":"10.1038\/modpathol.2016.108"},{"key":"e_1_2_13_22_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1470-2045(17)30624-1"},{"key":"e_1_2_13_23_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13569-016-0064-0"},{"key":"e_1_2_13_24_1","doi-asserted-by":"publisher","DOI":"10.1002\/cncr.32515"},{"key":"e_1_2_13_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejca.2021.03.039"},{"key":"e_1_2_13_26_1","doi-asserted-by":"publisher","DOI":"10.1002\/cncr.35628"},{"key":"e_1_2_13_27_1","unstructured":"National Center for Biotechnology Information of National Institute of Health \u201cTrial of Sunitinib and\/or Nivolumab Plus Chemotherapy in Advanced Soft Tissue and Bone Sarcomas (ImmunoSarc) \u201d2024 https:\/\/clinicaltrials.gov\/study\/NCT03277924."}],"container-title":["Cancer Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/cnr2.70426","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/cnr2.70426","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/cnr2.70426","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,3,8]],"date-time":"2026-03-08T16:16:29Z","timestamp":1772986589000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/cnr2.70426"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,1,29]]},"references-count":26,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2026,2]]}},"alternative-id":["10.1002\/cnr2.70426"],"URL":"https:\/\/doi.org\/10.1002\/cnr2.70426","archive":["Portico"],"relation":{},"ISSN":["2573-8348","2573-8348"],"issn-type":[{"value":"2573-8348","type":"print"},{"value":"2573-8348","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,1,29]]},"assertion":[{"value":"2024-05-20","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-11-27","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-01-29","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e70426"}}