{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,5,14]],"date-time":"2025-05-14T04:51:03Z","timestamp":1747198263606,"version":"3.40.5"},"reference-count":9,"publisher":"Walter de Gruyter GmbH","issue":"1","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,1,27]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Adrenocortical tumor (ACT) is a rare malignant tumor which usually present with Cushing syndrome and\u00a0virilization. Paraneoplastic syndromes (PNS) due to neoplasms can occur with peptides or cytokines secreted by the tumor. Here, we report a 13-month-old-male presented with severe masculinization. He had signs of precocious puberty with enlarged testicles, very high testosterone levels but low levels of gonadotrophins, and elevated \u03b2-hCG. He underwent a left nephrectomy. The histopathological evaluation revealed a diagnosis of adrenocortical neoplasm. The levels of androgens and \u03b2-hCG normalized after the resection of tumor, and the clinical findings improved within few months. We report the first pediatric patient with peripheral precocious puberty due to an ACT that secretes \u03b2-hCG as PNS. A\u00a0\u03b2-hCG secreting ACT can cause severe virilization due to\u00a0increased gonadal androgens stimulated by \u03b2-hCG as\u00a0well as due to increased adrenal androgens from the tumor.<\/jats:p>","DOI":"10.1515\/jpem-2022-0384","type":"journal-article","created":{"date-parts":[[2022,11,4]],"date-time":"2022-11-04T08:26:45Z","timestamp":1667550405000},"page":"96-100","source":"Crossref","is-referenced-by-count":1,"title":["Human chorionic gonadotrophin secreting adrenocortical neoplasm presenting with peripheral precocious puberty in an infant"],"prefix":"10.1515","volume":"36","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4880-8175","authenticated-orcid":false,"given":"Fatma","family":"Dursun","sequence":"first","affiliation":[{"name":"Division of Endocrinology, Department of Pediatrics , University of Health Sciences, Umraniye Training and Research Hospital , Istanbul , Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3473-5051","authenticated-orcid":false,"given":"Zekeriya","family":"\u0130l\u00e7e","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery , University of Health Sciences, Umraniye Training and Research Hospital , Istanbul , Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6655-1014","authenticated-orcid":false,"given":"\u0130lkay","family":"Tosun","sequence":"additional","affiliation":[{"name":"Department of Pathology , University of Health Sciences, Umraniye Training and Research Hospital , Istanbul , Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7945-730X","authenticated-orcid":false,"given":"P\u0131nar","family":"Atla","sequence":"additional","affiliation":[{"name":"Division of Endocrinology, Department of Pediatrics , University of Health Sciences, Umraniye Training and Research Hospital , Istanbul , Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9417-2847","authenticated-orcid":false,"given":"Sevin\u00e7","family":"Kal\u0131n","sequence":"additional","affiliation":[{"name":"Department of Pediatric Radiology , University of Health Sciences, Umraniye Training and Research Hospital , Istanbul , Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9464-9737","authenticated-orcid":false,"given":"Fatma Tu\u011fba","family":"G\u00fcven\u00e7","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery , University of Health Sciences, Umraniye Training and Research Hospital , Istanbul , Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0585-5123","authenticated-orcid":false,"given":"\u00dclk\u00fc Miray","family":"Y\u0131ld\u0131r\u0131m","sequence":"additional","affiliation":[{"name":"Division of Oncology, Department of Pediatrics , University of Health Sciences, Umraniye Training and Research Hospital , Istanbul , Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8663-3452","authenticated-orcid":false,"given":"Heves","family":"K\u0131rm\u0131z\u0131bekmez","sequence":"additional","affiliation":[{"name":"Division of Endocrinology, Department of Pediatrics , University of Health Sciences, Umraniye Training and Research Hospital , Istanbul , Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"374","published-online":{"date-parts":[[2022,11,7]]},"reference":[{"key":"2023033122552049639_j_jpem-2022-0384_ref_001","doi-asserted-by":"crossref","unstructured":"Pinto, EM, Zambetti, GP, Galindo, CR. Pediatric adrenocortical tumours. Best Pract Res Clin Endocrinol Metabol 2020;34:101448. https:\/\/doi.org\/10.1016\/j.beem.2020.101448.","DOI":"10.1016\/j.beem.2020.101448"},{"key":"2023033122552049639_j_jpem-2022-0384_ref_002","doi-asserted-by":"crossref","unstructured":"Gupta, N, Rivera, M, Novotny, P, Rodriguez, V, Bancos, I, Lteif, A. Adrenocortical carcinoma in children: a Clinicopathological analysis of 41 patients at the Mayo Clinic from 1950-2017. Horm Res Paediatr 2018;90:8\u201318. https:\/\/doi.org\/10.1159\/000488855.","DOI":"10.1159\/000488855"},{"key":"2023033122552049639_j_jpem-2022-0384_ref_003","doi-asserted-by":"crossref","unstructured":"Atay, Z, Yesilkaya, E, Erdeve, SS, Turan, S, Akin, L, Eren, E, et al.. The etiology and clinical features of non-CAH gonadotropin-independent precocious puberty: a multicenter study. 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Clin Biochem 2004;37:549\u201361. https:\/\/doi.org\/10.1016\/j.clinbiochem.2004.05.008.","DOI":"10.1016\/j.clinbiochem.2004.05.008"},{"key":"2023033122552049639_j_jpem-2022-0384_ref_006","doi-asserted-by":"crossref","unstructured":"Weiss, LM, Medeiros, LJ, Vickery, ALJr. Pathologic features of prognostic significance in adrenocortical carcinoma. Am J Surg Pathol 1989;13:202e6. https:\/\/doi.org\/10.1097\/00000478-198903000-00004.","DOI":"10.1097\/00000478-198903000-00004"},{"key":"2023033122552049639_j_jpem-2022-0384_ref_010","doi-asserted-by":"crossref","unstructured":"Alsakka, M, Ejeckam, GC, Homsi, UA, Sharara, H, Emadi, MI, Lopez, AC, et al.. Chorionic gonadotrophins-secreting adrenal cortical carcinoma: a mimicking a malignant trophoblastic disease. BJOG 1999;6:989\u201391. https:\/\/doi.org\/10.1111\/j.1471-0528.1999.tb08444.x.","DOI":"10.1111\/j.1471-0528.1999.tb08444.x"},{"key":"2023033122552049639_j_jpem-2022-0384_ref_008","doi-asserted-by":"crossref","unstructured":"Wendt, S, Shelso, J, Wright, K, Furman, W. Neoplastic causes of abnormal puberty. Pediatr Blood Cancer 2014;61:664\u201371. https:\/\/doi.org\/10.1002\/pbc.24825.","DOI":"10.1002\/pbc.24825"},{"key":"2023033122552049639_j_jpem-2022-0384_ref_009","doi-asserted-by":"crossref","unstructured":"Gattuso, G, Casanova, M, Biassoni, V, Terenziani, M, Schiavello, E, Sironi, G, et al.. Precocious pseudopuberty, a paraneoplastic\u00a0manifestation: a report of 2 cases. Tumori 2020;106:NP14\u201317. https:\/\/doi.org\/10.1177\/0300891620925532.","DOI":"10.1177\/0300891620925532"}],"container-title":["Journal of Pediatric Endocrinology and Metabolism"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.degruyter.com\/document\/doi\/10.1515\/jpem-2022-0384\/xml","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/www.degruyter.com\/document\/doi\/10.1515\/jpem-2022-0384\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,4,1]],"date-time":"2023-04-01T12:20:12Z","timestamp":1680351612000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.degruyter.com\/document\/doi\/10.1515\/jpem-2022-0384\/html"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,11,7]]},"references-count":9,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2022,11,23]]},"published-print":{"date-parts":[[2023,1,27]]}},"alternative-id":["10.1515\/jpem-2022-0384"],"URL":"https:\/\/doi.org\/10.1515\/jpem-2022-0384","relation":{},"ISSN":["0334-018X","2191-0251"],"issn-type":[{"type":"print","value":"0334-018X"},{"type":"electronic","value":"2191-0251"}],"subject":[],"published":{"date-parts":[[2022,11,7]]}}}