{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T16:03:21Z","timestamp":1791821001738,"version":"4.3.4"},"reference-count":27,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2026,1,19]],"date-time":"2026-01-19T00:00:00Z","timestamp":1768780800000},"content-version":"vor","delay-in-days":18,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,1,1]],"date-time":"2026-01-01T00:00:00Z","timestamp":1767225600000},"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":[[2026,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>CIC\u2010rearranged sarcomas (CRS) are rare tumors predominantly affecting young adults. Despite distinct biology, CRS are often treated like Ewing sarcomas with multi\u2010agent chemotherapy, surgery, and radiotherapy, although the benefits of each remain unclear. We report the impact of treatment on response and survival.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>This retrospective multicenter study included patients diagnosed with CRS between 2002 and 2023. Demographic, treatment, and outcome data were collected. Event\u2010free (EFS) and overall survival (OS) were estimated using Kaplan\u2013Meier analysis; risk factors were assessed via log\u2010rank tests and Cox regression. Treatment response was assessed using RECIST.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Among 27 patients (median age 21.5\u2009years; range 8\u201383), 14 (52%) had localized and 12 (44%) metastatic disease (1 unknown). In the localized group, 6 (43%) received chemotherapy (Response: 2 complete, CR; 1 partial, PR; 1 mixed, MR; 1 stable, SD; 1 progressive disease, PD); 5 (36%) died\u20143 (21%) had received chemotherapy. Among metastatic patients, 10 (83%) received chemotherapy (1 CR, 5 PR, 2 MR, 1 PD); 9 died (one without chemotherapy). Local control was achieved in 12\/14 (86%) localized and 7\/12 (56%) metastatic patients. Only surgical resection was associated with improved OS (HR 0.13;\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&lt;\u20090.01) and EFS (HR 0.26;\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&lt;\u20090.01), particularly with R0 resection (EFS HR 0.23;\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.02). At median follow\u2010up of 18\u2009months, 2\u2010year EFS was 56% vs. 8% (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.02) and OS was 67% vs. 23% (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.02) for localized vs. metastatic disease.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Complete resection is critical in CRS. Chemotherapy benefit remains uncertain, underscoring the need for improved local control and novel, biology\u2010driven therapies.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/cam4.71495","type":"journal-article","created":{"date-parts":[[2026,1,20]],"date-time":"2026-01-20T03:43:47Z","timestamp":1768880627000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["A Real\u2010World Analysis of Outcomes in CIC\u2010Rearranged Sarcomas: A Canadian Sarcoma Research and Clinical Collaboration (CanSaRCC) Study"],"prefix":"10.1002","volume":"15","author":[{"given":"Talya","family":"Wittmann Dayagi","sequence":"first","affiliation":[{"name":"Division of Haematology and Oncology, Department of Pediatrics, the Hospital for Sick Children University of Toronto  Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hagit","family":"Peretz Soroka","sequence":"additional","affiliation":[{"name":"Division of Haematology and Oncology, Department of Pediatrics, the Hospital for Sick Children University of Toronto  Toronto Ontario Canada"},{"name":"Division of Medical Oncology, Princess Margaret Cancer Centre University of Toronto  Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alannah","family":"Smrke","sequence":"additional","affiliation":[{"name":"Department of Medical Oncology, BC Cancer University of British Columbia  Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3315-4383","authenticated-orcid":false,"given":"Rebecca J.","family":"Deyell","sequence":"additional","affiliation":[{"name":"Division of Pediatric Hematology\/Oncology\/BMT, B.C. Children's Hospital and Research Institute University of British Columbia  Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiaolan","family":"Feng","sequence":"additional","affiliation":[{"name":"Division of Medical Oncology Tom Baker Cancer Center  Calgary Alberta Canada"},{"name":"Arthur Child Comprehensive Cancer Centre  Calgary Alberta Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6071-0042","authenticated-orcid":false,"given":"Sapna","family":"Oberoi","sequence":"additional","affiliation":[{"name":"Department of Pediatrics and Child Health, Max Rady College of Medicine University of Manitoba  Winnipeg Manitoba Canada"},{"name":"Department of Pediatric Hematology\u2010Oncology, CancerCare Manitoba University of Manitoba  Winnipeg Manitoba Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shantanu","family":"Banerji","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine, Rady Faculty of Medicine, CancerCare Manitoba University of Manitoba  Winnipeg Manitoba Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jonathan","family":"Noujaim","sequence":"additional","affiliation":[{"name":"Division of Medical Oncology, H\u00f4pital Maisonneuve Rosemont University of Montreal  Montreal Quebec Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicolas","family":"Prud'homme","sequence":"additional","affiliation":[{"name":"Division of Medical Oncology Centre Hospitalier Universitaire Sainte\u2010Justine  Montreal Quebec Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ramy","family":"Saleh","sequence":"additional","affiliation":[{"name":"Department of Medical Oncology, McGill University Health Center McGill University  Montreal Quebec Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4424-5360","authenticated-orcid":false,"given":"Omar Farooq","family":"Khan","sequence":"additional","affiliation":[{"name":"Division of Medical Oncology, Department of Oncology, Cumming School of Medicine University of Calgary  Calgary Alberta Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9432-2390","authenticated-orcid":false,"given":"Jonathan Willard","family":"Bush","sequence":"additional","affiliation":[{"name":"Department of Pathology and Laboratory Medicine, BC Children's Hospital University of British Columbia  Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bilal","family":"Marwa","sequence":"additional","affiliation":[{"name":"Department of Pediatrics and Oncology University of Saskatchewan, Saskatchewan Cancer Agency, Saskatchewan Health Authority  Saskatoon Saskatchewan Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1962-7893","authenticated-orcid":false,"given":"Geoffrey","family":"Watson","sequence":"additional","affiliation":[{"name":"Division of Medical Oncology, Princess Margaret Cancer Centre University of Toronto  Toronto Ontario Canada"},{"name":"Department of Medical Oncology Mount Sinai Hospital  Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Caroline","family":"Holloway","sequence":"additional","affiliation":[{"name":"Division of Radiation Oncology, BC Cancer University of British Columbia  Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lingxin","family":"Zhang","sequence":"additional","affiliation":[{"name":"Department of Pathology and Laboratory Medicine, Mount Sinai Hospital University of Toronto  Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0825-5070","authenticated-orcid":false,"given":"Abha Anshu","family":"Gupta","sequence":"additional","affiliation":[{"name":"Division of Haematology and Oncology, Department of Pediatrics, the Hospital for Sick Children University of Toronto  Toronto Ontario Canada"},{"name":"Division of Medical Oncology, Princess Margaret Cancer Centre University of Toronto  Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9475-0077","authenticated-orcid":false,"given":"Jack","family":"Brzezinski","sequence":"additional","affiliation":[{"name":"Division of Haematology and Oncology, Department of Pediatrics, the Hospital for Sick Children University of Toronto  Toronto Ontario Canada"},{"name":"Cancer and Blood Disorders Center Seattle Children's Hospital  Seattle Washington USA"},{"name":"Department of Pediatrics University of Washington  Seattle Washington USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,1,19]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00330-008-1292-3"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pcl.2014.09.012"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0000000000001552"},{"key":"e_1_2_10_5_1","first-page":"70","article-title":"The 2020 WHO Classification of Soft Tissue Tumours: News and Perspectives","volume":"113","author":"Angelo Dei Tos C. 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