{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,25]],"date-time":"2026-08-25T15:30:45Z","timestamp":1787671845350,"version":"build-2736575974"},"reference-count":25,"publisher":"Wiley","issue":"10","license":[{"start":{"date-parts":[[2026,8,8]],"date-time":"2026-08-08T00:00:00Z","timestamp":1786147200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,8,8]],"date-time":"2026-08-08T00:00:00Z","timestamp":1786147200000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100002002","name":"Cancer Research Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100002002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100006058","name":"St. Baldrick's Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100006058","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100016610","name":"Andrew McDonough B+ Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100016610","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100018762","name":"Seattle Children's Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100018762","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100003287","name":"Rally Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100003287","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100001411","name":"King Baudouin Foundation United States","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100001411","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pediatric Blood &amp;amp; Cancer"],"published-print":{"date-parts":[[2026,10]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Patients with orbital rhabdomyosarcoma (ORMS) historically have 10\u2010year overall survival (OS) rates &gt; 85% with multimodal therapy. We aimed to determine if favorable outcomes were preserved on contemporary international cooperative group trials in the context of shifting treatment approaches, including chemotherapy reduction and changes in the indications for radiation therapy (RT) and RT dose.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>We identified patients with newly diagnosed ORMS (orbit and eyelid sites) from 11 North American and European cooperative group trials from 1991 to 2016. Demographic and clinical characteristics were collected. Event\u2010free survival (EFS) and OS were calculated from the time of trial enrollment except post\u2010relapse OS, which was calculated from the time of recurrence.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Of 679 patients with ORMS, the 10\u2010year EFS and OS were 74.6% (71.0%\u201377.8%) and 92.9% (90.6%\u201394.8%). In patients with Group III, non\u2010alveolar ORMS (\n                      <jats:italic>n<\/jats:italic>\n                      = 425, 62.6%), the 10\u2010year EFS and OS were 78% (74%\u201382%) and 95% (92%\u201397%), respectively. Anaplasia was associated with inferior 10\u2010year EFS (79.8%\u00a0vs. 84.2%,\n                      <jats:italic>p<\/jats:italic>\n                      = 0.03), and the 10\u2010year post\u2010recurrence OS was 78% (67%\u201386%). Patients with localized alveolar ORMS (\n                      <jats:italic>n<\/jats:italic>\n                      = 81; PAX::FOXO1 fusion positive in 13\/33 patients with known fusion status) had a 10\u2010year EFS of 71% (60%\u201380%) and OS of 88% (79%\u201394%). Infants &lt; 1 year (\n                      <jats:italic>n<\/jats:italic>\n                      = 22) had a 5\u2010year EFS of 31% (12%\u201352%) and OS of 61% (37%\u201378%). Metastatic ORMS (age \u22651) was rare (\n                      <jats:italic>n<\/jats:italic>\n                      = 5; 4\/5 alveolar histology), and no patients survived more than 4 years.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Favorable survival in patients with ORMS was preserved despite shifts in treatment strategies. Patients with localized alveolar ARMS and recurrent ORMS had favorable survival. Infants &lt; 1 year and those \u2265 1 with metastatic disease had poor prognosis and require new therapeutic approaches.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/1545-5017.70613","type":"journal-article","created":{"date-parts":[[2026,8,8]],"date-time":"2026-08-08T13:24:18Z","timestamp":1786195458000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Outcomes of Children With Orbital Rhabdomyosarcoma, 1991\u20132016: A Report From the International Soft Tissue Sarcoma Consortium (INSTRuCT)"],"prefix":"10.1002","volume":"73","author":[{"given":"Jonathan","family":"Metts","sequence":"first","affiliation":[{"name":"Sarcoma Department Moffitt Cancer Center  Tampa Florida USA"},{"name":"Cancer and Blood Disorders Institute Johns Hopkins All Children's Hospital  St Petersburg Florida USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James","family":"Anderson","sequence":"additional","affiliation":[{"name":"University of Nebraska Medical Center  Omaha Nebraska USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christian","family":"Vokuhl","sequence":"additional","affiliation":[{"name":"Section of Pediatric Pathology Department of Pathology University Hospital Bonn  Bonn Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ewa","family":"Koscielniak","sequence":"additional","affiliation":[{"name":"Klinik F\u00fcr Kinder\u2010 und Jugendmedizin Kinderheilkunde I \u2010 Allgemeine P\u00e4diatrie H\u00e4matologie Und Onkologie Universit\u00e4tsklinikum T\u00fcbingen  T\u00fcbingen Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cristian","family":"Urla","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery and Pediatric Urology University Children's Hospital Eberhard Karls University T\u00fcbingen  T\u00fcbingen Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rajkumar","family":"Venkatramani","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Texas Children's Cancer and Hematology Centers Baylor College of Medicine  Houston Texas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"John","family":"Breneman","sequence":"additional","affiliation":[{"name":"Department of Radiation Oncology University of Cincinnati College of Medicine  Cincinnati Ohio USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Julie","family":"Bradley","sequence":"additional","affiliation":[{"name":"Department of Radiation Oncology University of Florida College of Medicine  Jacksonville Florida USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael","family":"Arnold","sequence":"additional","affiliation":[{"name":"Department of Pathology and Laboratory Medicine Children's Hospital Colorado  Aurora Colorado USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Veronique","family":"Minard\u2010Colin","sequence":"additional","affiliation":[{"name":"Department of Pediatric and Adolescent Oncology Gustave Roussy Cancer Campus University Paris Saclay  Villejuif France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gianni","family":"Bisogno","sequence":"additional","affiliation":[{"name":"Department of Women's and Children's Health University of Padua  Padua Italy"},{"name":"Pediatric Hematology Oncology Division University Hospital of Padua  Padua Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Henry","family":"Mandeville","sequence":"additional","affiliation":[{"name":"The Royal Marsden Hospital and The Institute of Cancer Research  Surrey UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Johannes","family":"Merks","sequence":"additional","affiliation":[{"name":"Princess M\u00e1xima Center For Pediatric Oncology  Utrecht the Netherlands"},{"name":"Division of Imaging and Oncology University Medical Center Utrecht Utrecht University  Utrecht the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Beate","family":"Timmerman","sequence":"additional","affiliation":[{"name":"Department of Particle Therapy University Hospital Essen West German Proton Therapy Centre Essen (WPE) West German Cancer Center (WTZ)  Essen Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thekla","family":"von Kalle","sequence":"additional","affiliation":[{"name":"Klinikum Stuttgart \u2013 Olgahospital Stuttgart Cancer Centre Institute of Radiology  Stuttgart Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Monika","family":"Sparber\u2010Sauer","sequence":"additional","affiliation":[{"name":"Klinik F\u00fcr Kinder\u2010 und Jugendmedizin Kinderheilkunde I \u2010 Allgemeine P\u00e4diatrie H\u00e4matologie Und Onkologie Universit\u00e4tsklinikum T\u00fcbingen  T\u00fcbingen Germany"},{"name":"Stuttgart Cancer Center Zentrum F\u00fcr Kinder\u2010 und Jugend Frauenmedizin, Klinikum Stuttgart  Stuttgart Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David","family":"Walterhouse","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Ann &amp; Robert H. Lurie Children's Hospital of Chicago Northwestern University Feinberg School of Medicine Chicago Illinois USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,8,8]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41572\u2010018\u20100051\u20102"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.29511"},{"key":"e_1_2_11_4_1","doi-asserted-by":"publisher","DOI":"10.3390\/cancers17040615"},{"key":"e_1_2_11_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.clindermatol.2024.01.012"},{"key":"e_1_2_11_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.wneu.2024.07.110"},{"key":"e_1_2_11_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.prro.2019.10.002"},{"key":"e_1_2_11_8_1","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.2001.19.1.197"},{"key":"e_1_2_11_9_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.24289"},{"key":"e_1_2_11_10_1","doi-asserted-by":"publisher","DOI":"10.1002\/cncr.35743"},{"key":"e_1_2_11_11_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.29924"},{"key":"e_1_2_11_12_1","doi-asserted-by":"publisher","DOI":"10.1200\/CCI.21.00075"},{"key":"e_1_2_11_13_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.24679"},{"key":"e_1_2_11_14_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.31436"},{"key":"e_1_2_11_15_1","doi-asserted-by":"publisher","DOI":"10.1002\/cncr.35497"},{"key":"e_1_2_11_16_1","doi-asserted-by":"publisher","DOI":"10.1111\/aos.14596"},{"key":"e_1_2_11_17_1","doi-asserted-by":"publisher","DOI":"10.1002\/cncr.23929"},{"key":"e_1_2_11_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejca.2020.10.018"},{"key":"e_1_2_11_19_1","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.20.03060"},{"key":"e_1_2_11_20_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.26061"},{"key":"e_1_2_11_21_1","doi-asserted-by":"publisher","DOI":"10.1080\/0284186X.2019.1627412"},{"key":"e_1_2_11_22_1","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.2005.05.3801"},{"key":"e_1_2_11_23_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.25862"},{"key":"e_1_2_11_24_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.24532"},{"key":"e_1_2_11_25_1","doi-asserted-by":"publisher","DOI":"10.1002\/cncr.25887"},{"key":"e_1_2_11_26_1","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.2015.63.4048"}],"container-title":["Pediatric Blood &amp; Cancer"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/1545-5017.70613","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/1545-5017.70613","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/1545-5017.70613","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,25]],"date-time":"2026-08-25T15:00:22Z","timestamp":1787670022000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/1545-5017.70613"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,8,8]]},"references-count":25,"journal-issue":{"issue":"10","published-print":{"date-parts":[[2026,10]]}},"alternative-id":["10.1002\/1545-5017.70613"],"URL":"https:\/\/doi.org\/10.1002\/1545-5017.70613","archive":["Portico"],"relation":{},"ISSN":["1545-5009","1545-5017"],"issn-type":[{"value":"1545-5009","type":"print"},{"value":"1545-5017","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,8,8]]},"assertion":[{"value":"2026-04-09","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-07-23","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-08-08","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e70613"}}