{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,22]],"date-time":"2026-09-22T13:15:56Z","timestamp":1790082956846,"version":"4.0.1"},"reference-count":27,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2024,1,23]],"date-time":"2024-01-23T00:00:00Z","timestamp":1705968000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2024,1,23]],"date-time":"2024-01-23T00:00:00Z","timestamp":1705968000000},"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":["Pediatric Blood &amp;amp; Cancer"],"published-print":{"date-parts":[[2024,4]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Rationale<\/jats:title>\n                    <jats:p>Sex cord\u2010stromal tumors (SCST) are hormonally active and rare. The aim was to describe their endocrinological presentation and outcomes.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Method<\/jats:title>\n                    <jats:p>Patients (&lt;\u00a019 years) registered in the TGM13 registry between 2014 and 2021 for SCST were selected.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Sixty\u2010three ovarian SCST (juvenile granulosa tumor (JGT)\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u00a034, Sertoli\u2010Leydig cell tumor (SLCT)\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u00a017, other SCST\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u00a012) were included. Median age was 13.1 years (0.4\u201017.4). Germline\n                      <jats:italic>DICER1<\/jats:italic>\n                      pathogenic variant was present in 9\/17 SLCT. Sixty\u2010one were FIGO stage I (IC\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u00a014). Adjuvant chemotherapy was administered for 15. Seven had recurrence (FIGO IA\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u00a03, IX\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u00a02, III\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u00a02), leading to one death. With a median follow\u2010up of 42 months (2.5\u201092), the 3\u2010year progression\u2010free survival (PFS) was 89% (95% CI 76%\u201095%). Median age was 6.4 years (0.1\u201012.9) among the 15 testicular SCST (Leydig cell tumor\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u00a06, JGT\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u00a05, Sertoli cell tumor\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u00a03, mixed SCST\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u00a01). Tumor\u2010nodes\u2010metastases (TNM) stage was pSI in 14. Eight underwent a tumorectomy, 7 an orchiectomy. None experienced recurrence.\n                    <\/jats:p>\n                    <jats:p>Endocrinological data were reviewed for 41 patients (18 prepubescent). Endocrine symptoms were present at diagnosis in 29\/34 females and 2\/7 males (gynecomastia). After a median follow\u2010up of 11 months, 15 patients had persistent endocrine abnormalities: gynecomastia\/breast growth (2 males, 1 prepubescent female), precocious\/advanced puberty (4 prepubescent females), and hirsutism\/menstruation disorders\/voice hoarseness\/hot flashes (8 pubescent females). The mean height at the last follow\u2010up was within normal ranges (+0.3 standard deviation).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>SCSTs have a favorable prognosis. Tumorectomy appears safe with testicular primary. Endocrinological disorders, common at diagnosis, may persist warranting endocrinological follow\u2010up.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/pbc.30864","type":"journal-article","created":{"date-parts":[[2024,1,23]],"date-time":"2024-01-23T08:37:38Z","timestamp":1705999058000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Oncological and endocrinological outcomes for children and adolescents with testicular and ovarian sex cord\u2010stromal tumors. Results of the TGM13 National Registry"],"prefix":"10.1002","volume":"71","author":[{"ORCID":"https:\/\/orcid.org\/0009-0004-7948-9220","authenticated-orcid":false,"given":"Clemence","family":"Fuentes","sequence":"first","affiliation":[{"name":"Department of Pediatric Oncology Institut d'Hemato\u2010oncologie Pediatrique  Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yaelle","family":"Ouldbey","sequence":"additional","affiliation":[{"name":"Statistical Unit Clinical Research Department Centre L\u00e9on B\u00e9rard  Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2520-139X","authenticated-orcid":false,"given":"Daniel","family":"Orbach","sequence":"additional","affiliation":[{"name":"SIREDO Oncology Center (Care, Innovation and Research for Children, Adolescents and Young Adults with Cancer) Institut Curie PSL University  Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1991-3585","authenticated-orcid":false,"given":"Helene","family":"Sudour\u2010Bonnange","sequence":"additional","affiliation":[{"name":"Anti\u2010Cancer Center Oscar Lambret Pediatric and Adolescents Oncology Unit  Lille France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cecile","family":"Verit\u00e9","sequence":"additional","affiliation":[{"name":"Pediatric Hematology\u2010Oncology Unit Pellegrin Hospital, CHU Bordeaux  Bordeaux France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Angelique","family":"Rome","sequence":"additional","affiliation":[{"name":"Department of Pediatric Oncology of Timone Children's Hospital  Marseille France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cecile","family":"Dumesnil","sequence":"additional","affiliation":[{"name":"Department of Pediatric Oncology University Hospital Center of Rouen  Rouen France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Estelle","family":"Thebaud","sequence":"additional","affiliation":[{"name":"Department of Pediatric Oncology University Hospital Center of Nantes  Nantes France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frederic","family":"Hameury","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery H\u00f4pital Femme M\u00e8re Enfant  Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frederique","family":"Dijoud","sequence":"additional","affiliation":[{"name":"Institut Multisite de Pathologie CHU de Lyon  Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sylvie","family":"Chabaud","sequence":"additional","affiliation":[{"name":"Statistical Unit Clinical Research Department Centre L\u00e9on B\u00e9rard  Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M. Daval","family":"Cote","sequence":"additional","affiliation":[{"name":"D\u00e9partement of Endocrinology and Pediatric Diabetology Hospices civils de Lyon  Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7603-7828","authenticated-orcid":false,"given":"Brice","family":"Fresneau","sequence":"additional","affiliation":[{"name":"Gustave Roussy, Universit\u00e9 Paris\u2010Saclay, Department of Children and Adolescent Oncology Paris\u2010Saclay University, Paris\u2010Sud University CESP, INSERM  Villejuif France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4717-4943","authenticated-orcid":false,"given":"Cecile","family":"Faure\u2010Conter","sequence":"additional","affiliation":[{"name":"Department of Pediatric Oncology Institut d'Hemato\u2010oncologie Pediatrique  Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2024,1,23]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bulcan.2015.04.012"},{"key":"e_1_2_10_3_1","volume-title":"Female genital tumours WHO classification of tumours","author":"WHO Classification of Tumours Editorial Board","year":"2020"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.1111\/his.14675"},{"key":"e_1_2_10_5_1","doi-asserted-by":"publisher","DOI":"10.1038\/modpathol.3800311"},{"key":"e_1_2_10_6_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa1102903"},{"key":"e_1_2_10_7_1","doi-asserted-by":"publisher","DOI":"10.1586\/14737159.3.4.471"},{"key":"e_1_2_10_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.29017"},{"key":"e_1_2_10_9_1","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.2007.11.1005"},{"key":"e_1_2_10_10_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.22918"},{"issue":"9","key":"e_1_2_10_11_1","first-page":"793","article-title":"Granulosa cell tumors of the ovary in children and adolescents. 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