{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,8]],"date-time":"2026-07-08T05:58:37Z","timestamp":1783490317198,"version":"3.55.0"},"reference-count":36,"publisher":"Walter de Gruyter GmbH","issue":"9","funder":[{"name":"Mitteldeutschen Kinderkrebsforschung","award":["not applicable"],"award-info":[{"award-number":["not applicable"]}]},{"DOI":"10.13039\/501100007311","name":"German Childhood Cancer Foundation","doi-asserted-by":"crossref","award":["DKS 2014.06, DKS 2017.16, DKS 2021.11, DKS 2024.16"],"award-info":[{"award-number":["DKS 2014.06, DKS 2017.16, DKS 2021.11, DKS 2024.16"]}],"id":[{"id":"10.13039\/501100007311","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Faculty of Medicine, University of Augsburg","award":["not applicable"],"award-info":[{"award-number":["not applicable"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,9,25]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec id=\"j_jpem-2025-0198_abs_001\">\n                  <jats:title>Objectives<\/jats:title>\n                  <jats:p>To explore the clinical spectrum and symptom duration in pediatric adrenocortical tumors (pACTs), with a focus on identifying cases that may reflect gradual tumor progression.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_jpem-2025-0198_abs_002\">\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>We retrospectively analyzed data from 110 pediatric patients with pACTs enrolled in the German Pediatric Oncology Hematology-Malignant Endocrine Tumor (GPOH-MET) studies (1997\u20132022). Endocrine symptom duration, histopathological classification, and clinical outcomes were assessed. Patients with symptom durations\u00a0\u22652 standard deviations (SDs) from the mean were defined as outliers and evaluated for potential progression.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_jpem-2025-0198_abs_003\">\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>The cohort included 31 patients with adrenocortical adenomas (ACAs), 12 with tumors of uncertain malignant potential (ACx), and 67 with adrenocortical carcinomas (ACCs). Seven patients (6.4\u202f%) showed markedly prolonged symptom duration, including four with ACC. One representative case demonstrated a nearly 5-year course from initial androgen excess to metastatic ACC, with evolving biochemical features and a diagnostic urinary steroid profile indicative of adrenal tumor activity.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_jpem-2025-0198_abs_004\">\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>A small subset of pACTs may present with prolonged endocrine symptoms, possibly reflecting gradual tumor evolution. While molecular validation is lacking, these findings support the need for early recognition and further research into the natural history of pACTs.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1515\/jpem-2025-0198","type":"journal-article","created":{"date-parts":[[2025,6,27]],"date-time":"2025-06-27T17:55:47Z","timestamp":1751046947000},"page":"931-938","source":"Crossref","is-referenced-by-count":1,"title":["Prolonged symptom duration and the potential for gradual progression in pediatric adrenocortical tumors: observations from the MET studies"],"prefix":"10.1515","volume":"38","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4577-0503","authenticated-orcid":false,"given":"Michaela","family":"Kuhlen","sequence":"first","affiliation":[{"name":"Pediatrics and Adolescent Medicine, Faculty of Medicine , 531257 University of Augsburg , Augsburg , Germany"},{"name":"Bavarian Cancer Research Center (BZKF) , Augsburg , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stefan A.","family":"Wudy","sequence":"additional","affiliation":[{"name":"Steroid Research & Mass Spectrometry Unit, Laboratory for Translational Hormone Analytics, Pediatric Endocrinology & Diabetology, Centre of Child and Adolescent Medicine , Justus Liebig University Giessen , Giessen , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Clara","family":"Baumann","sequence":"additional","affiliation":[{"name":"Department of Pediatric Kidney, Liver, Metabolic and Neurological Diseases , Children\u2019s Hospital, Hannover Medical School , Hannover , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4138-4536","authenticated-orcid":false,"given":"Christian","family":"Vokuhl","sequence":"additional","affiliation":[{"name":"Section of Pediatric Pathology , Institute of Pathology, University of Bonn , Bonn , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7232-9532","authenticated-orcid":false,"given":"Michaela F.","family":"Hartmann","sequence":"additional","affiliation":[{"name":"Steroid Research & Mass Spectrometry Unit, Laboratory for Translational Hormone Analytics, Pediatric Endocrinology & Diabetology, Centre of Child and Adolescent Medicine , Justus Liebig University Giessen , Giessen , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2672-4045","authenticated-orcid":false,"given":"Marina","family":"Kunstreich","sequence":"additional","affiliation":[{"name":"Pediatrics and Adolescent Medicine, Faculty of Medicine , 531257 University of Augsburg , Augsburg , Germany"},{"name":"Department of Pediatrics, Pediatric Hematology\/Oncology , Otto-von-Guericke-University , Magdeburg , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2617-8766","authenticated-orcid":false,"given":"Rainer","family":"Claus","sequence":"additional","affiliation":[{"name":"Bavarian Cancer Research Center (BZKF) , Augsburg , Germany"},{"name":"Pathology, Faculty of Medicine, University of Augsburg , Augsburg , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1732-1869","authenticated-orcid":false,"given":"Antje","family":"Redlich","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Pediatric Hematology\/Oncology , Otto-von-Guericke-University , Magdeburg , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"374","published-online":{"date-parts":[[2025,6,30]]},"reference":[{"key":"2025091715550578234_j_jpem-2025-0198_ref_001","doi-asserted-by":"crossref","unstructured":"Siegel, DA, King, J, Tai, E, Buchanan, N, Ajani, UA, Li, J. 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