{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,9]],"date-time":"2026-07-09T21:37:01Z","timestamp":1783633021746,"version":"3.55.0"},"reference-count":62,"publisher":"Oxford University Press (OUP)","issue":"6","license":[{"start":{"date-parts":[[2022,10,26]],"date-time":"2022-10-26T00:00:00Z","timestamp":1666742400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/pages\/standard-publication-reuse-rights"}],"content-domain":{"domain":["eje.bioscientifica.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2022,12,1]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Objective<\/jats:title><jats:p>Pediatric adrenocortical carcinoma (pACC) is rare and prognostic stratification remains challenging. We summarized the clinical prognostic factors of pACC and determined the prognostic value of the pediatric scoring system (pS-GRAS) in adaption to the recommendation (S-GRAS) of the European Network for the Study of Adrenal Tumors for the classification of adult ACC.<\/jats:p><\/jats:sec><jats:sec><jats:title>Design<\/jats:title><jats:p>Analysis of pACC patients of 33 available retrospective studies in the literature.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>We searched the PubMed and Embase databases for manuscripts regarding pACC. The pS-GRAS score was calculated as a sum of tumor stage (1\u2009=\u20090; 2\u20133\u2009=\u20091; 4\u2009=\u20092 points), grade (Ki67 index\/rate of mitosis 0\u20139%\/low\u2009=\u20090; 10\u201319%\/intermediate\u2009=\u20091; \u226520%\/high\u2009=\u20092 points), resection status (R0\u2009=\u20090; RX\u2009=\u20091; R1\u2009=\u20092; R2\u2009=\u20093 points), age (&amp;lt;4 years\u2009=\u20090; \u22654 years\u2009=\u20091 point), hormone-related symptoms (androgen production\u2009=\u20090; glucocorticoid\/mixed\/no hormone production\u2009=\u20091 point) generating 10 scores and 4 groups (1: 0\u20132, 2: 3\u20134, 3: 5, 4: 6\u20139). The primary endpoint was overall survival (OS).<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>We included 733 patients. The median age was 2.5 years and &amp;gt;85% of pACC showed hormone activity (mixed 50%, androgen 29%, glucocorticoid 21%). Androgen production was associated with a superior OS. Increasing age correlated with higher rates of inactive or only glucocorticoid-producing tumors, advanced tumor stage, and case fatality. Especially infants &amp;lt; 4 years showed more often low-risk constellations with an increased OS for all tumor stages. The pS-GRAS score correlated with clinical outcome; median OS was 133 months (95% CI: 36\u2013283) in group 1 (n\u2009=\u200949), 110 months (95% CI: 2.9\u2013314) in group 2 (n\u2009=\u200957), 49 months (95% CI: 5.8\u2013278) in group 3 (n\u2009=\u200918), and 16 months (95% CI: 2.4\u2013267) in group 4; (n\u2009=\u200911) P\u2009&amp;lt;\u20090.05).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>The pS-GRAS score seems to have a high predictive value in the pACC patients, may serve as a helpful tool for risk stratification in future studies, and should be evaluated prospectively in an international context.<\/jats:p><\/jats:sec>","DOI":"10.1530\/eje-22-0173","type":"journal-article","created":{"date-parts":[[2022,10,4]],"date-time":"2022-10-04T09:21:18Z","timestamp":1664875278000},"page":"751-763","update-policy":"https:\/\/doi.org\/10.1530\/crossmarkpolicy-7","source":"Crossref","is-referenced-by-count":14,"title":["Assessment of prognostic factors in pediatric adrenocortical tumors: a systematic review and evaluation of a modified S-GRAS score"],"prefix":"10.1093","volume":"187","author":[{"given":"Maria","family":"Riedmeier","sequence":"first","affiliation":[{"name":"Division of Pediatric Hematology, Oncology and Stem Cell Transplantation, Department of Pediatrics, University Hospital, University of Wuerzburg , Wuerzburg, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Boris","family":"Decarolis","sequence":"additional","affiliation":[{"name":"Department of Pediatric Oncology and Hematology, University Children's Hospital of Cologne, Medical Faculty , Cologne, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Imme","family":"Haubitz","sequence":"additional","affiliation":[{"name":"Division of Pediatric Hematology, Oncology and Stem Cell Transplantation, Department of Pediatrics, University Hospital, University of Wuerzburg , Wuerzburg, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joachim","family":"Reibetanz","sequence":"additional","affiliation":[{"name":"Department of General, Visceral, Transplantation, Vascular and Pediatric Surgery, University Hospital, University of Wuerzburg , Wuerzburg, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7777-0909","authenticated-orcid":false,"given":"Armin","family":"Wiegering","sequence":"additional","affiliation":[{"name":"Department of General, Visceral, Transplantation, Vascular and Pediatric Surgery, University Hospital, University of Wuerzburg , Wuerzburg, Germany"},{"name":"Department of Biochemistry and Molecular Biology, University of Wuerzburg , Wuerzburg, Germany"},{"name":"Comprehensive Cancer Centre Mainfranken, University of Wuerzburg Medical Centre , Wuerzburg, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christoph","family":"H\u00e4rtel","sequence":"additional","affiliation":[{"name":"Division of Pediatric Hematology, Oncology and Stem Cell Transplantation, Department of Pediatrics, University Hospital, University of Wuerzburg , Wuerzburg, Germany"},{"name":"Comprehensive Cancer Centre Mainfranken, University of Wuerzburg Medical Centre , Wuerzburg, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paul-Gerhardt","family":"Schlegel","sequence":"additional","affiliation":[{"name":"Division of Pediatric Hematology, Oncology and Stem Cell Transplantation, Department of Pediatrics, University Hospital, University of Wuerzburg , Wuerzburg, Germany"},{"name":"Comprehensive Cancer Centre Mainfranken, University of Wuerzburg Medical Centre , Wuerzburg, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6170-6398","authenticated-orcid":false,"given":"Martin","family":"Fassnacht","sequence":"additional","affiliation":[{"name":"Comprehensive Cancer Centre Mainfranken, University of Wuerzburg Medical Centre , Wuerzburg, Germany"},{"name":"Division of Endocrinology and Diabetes, Department of Medicine, University Hospital, University of Wuerzburg , Wuerzburg, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2802-4034","authenticated-orcid":false,"given":"Verena","family":"Wiegering","sequence":"additional","affiliation":[{"name":"Division of Pediatric Hematology, Oncology and Stem Cell Transplantation, Department of Pediatrics, University Hospital, University of Wuerzburg , Wuerzburg, Germany"},{"name":"Comprehensive Cancer Centre Mainfranken, University of Wuerzburg Medical Centre , Wuerzburg, 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