{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,9]],"date-time":"2026-09-09T23:18:23Z","timestamp":1788995903567,"version":"build-2803163510"},"reference-count":22,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2017,12,29]],"date-time":"2017-12-29T00:00:00Z","timestamp":1514505600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Pediatric Blood &amp; Cancer"],"published-print":{"date-parts":[[2018,4]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Purpose<\/jats:title><jats:p>In this report, we characterize the timing and behavior of malignant ovarian germ cell tumors (GCTs) in pediatric patients with dysgenetic gonads compared to those with normal gonadal development.<\/jats:p><\/jats:sec><jats:sec><jats:title>Patients and methods<\/jats:title><jats:p>Patients from the Children's Oncology Group AGCT0132 with malignant ovarian GCTs were included. Within this population, we sought to identify patients with gonadoblastoma, streak ovaries, or other evidence of gonadal dysgenesis (GD). Patients with malignant GCTs containing one or more of the following histologies\u2014yolk sac tumor, embryonal carcinoma, or choriocarcinoma\u2014were included. Patients were compared with respect to event\u2010free survival (EFS) and overall survival (OS).<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Nine patients with GD, including seven with gonadoblastoma (mean age, 9.3 years), were compared to 100 non\u2010GD patients (mean age, 12.1 years). The estimated 3\u2010year EFS for patients with GD was 66.7% (95% CI 28.2\u201387.8%) and for non\u2010GD patients was 88.8% (95% CI 80.2\u201393.8%). The estimated 3\u2010year OS for patients with GD was 87.5% (95% CI 38.7\u201398.1%) and for non\u2010GD patients was 97.6% (95% CI of 90.6\u201399.4%).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Patients presenting with nongerminomatous malignant ovarian GCTs in the context of GD have a higher rate of events and death than counterparts with normal gonads. These findings emphasize the importance of noting a contralateral streak ovary or gonadoblastoma at histology for any ovarian GCT and support the recommendation for early bilateral gonadectomy in patients known to have GD with Y chromosome material. In contrast to those with pure dysgerminoma, these patients may represent a high\u2010risk group that requires a more aggressive chemotherapy regimen.<\/jats:p><\/jats:sec>","DOI":"10.1002\/pbc.26913","type":"journal-article","created":{"date-parts":[[2017,12,29]],"date-time":"2017-12-29T15:26:41Z","timestamp":1514561201000},"source":"Crossref","is-referenced-by-count":21,"title":["Gonadal dysgenesis is associated with worse outcomes in patients with ovarian nondysgerminomatous tumors: A report of the Children's Oncology Group AGCT 0132 study"],"prefix":"10.1002","volume":"65","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5058-5973","authenticated-orcid":false,"given":"Bryan J.","family":"Dicken","sequence":"first","affiliation":[{"name":"Stollery Children's Hospital University of Alberta Hospital  Edmonton Alberta Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Deborah F.","family":"Billmire","sequence":"additional","affiliation":[{"name":"Riley Hospital for Children  Indianapolis Indiana"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mark","family":"Krailo","sequence":"additional","affiliation":[{"name":"Keck School of Medicine University of Southern California  Los Angeles California"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Caihong","family":"Xia","sequence":"additional","affiliation":[{"name":"Keck School of Medicine University of Southern California  Los Angeles California"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Furqan","family":"Shaikh","sequence":"additional","affiliation":[{"name":"The Hospital for Sick Children  Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"John W.","family":"Cullen","sequence":"additional","affiliation":[{"name":"Rocky Mountain Hospital for Children\u2010Presbyterian St Luke's Medical Center  Denver Colorado"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas A.","family":"Olson","sequence":"additional","affiliation":[{"name":"Children's Healthcare of Atlanta Emory University  Atlanta Georgia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Farzana","family":"Pashankar","sequence":"additional","affiliation":[{"name":"Yale University  New Haven Connecticut"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marcio H.","family":"Malogolowkin","sequence":"additional","affiliation":[{"name":"University of California Davis  Davis California"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James F.","family":"Amatruda","sequence":"additional","affiliation":[{"name":"University of Texas Southwestern Medical Center  Dallas Texas"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frederick J.","family":"Rescorla","sequence":"additional","affiliation":[{"name":"Riley Hospital for Children  Indianapolis Indiana"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rachel A.","family":"Egler","sequence":"additional","affiliation":[{"name":"Rainbow Babies and Children's Hospital  Cleveland Ohio"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jonathan H.","family":"Ross","sequence":"additional","affiliation":[{"name":"Rainbow Babies and Children's Hospital  Cleveland Ohio"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carlos","family":"Rodriguez\u2010Galindo","sequence":"additional","affiliation":[{"name":"St. Jude Children's Research Hospital  Memphis Tennessee"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A. 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