{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,16]],"date-time":"2026-10-16T10:23:45Z","timestamp":1792146225686,"version":"4.4.0"},"reference-count":29,"publisher":"Oxford University Press (OUP)","issue":"4","content-domain":{"domain":["eje.bioscientifica.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2010,4]]},"abstract":"<jats:sec><jats:title>Context<\/jats:title><jats:p>Only occasionally, endocrine-active tumors develop directly from hepatic tissue, and may lead to paraneoplastic syndromes (PNS). PNS mostly accompany malignancy of adulthood and are exceedingly rare in children.<\/jats:p><\/jats:sec><jats:sec><jats:title>Patient<\/jats:title><jats:p>A girl aged 6 years and 9 months presented with a 2-month history of rapidly progressive weight gain, abdominal distension, and polyuria\/pollakiuria accompanied by short episodes of abdominal pain. She showed the typical clinical features of Cushing's syndrome and a huge hepatic mass. An abdominal computed tomography (CT) scan revealed a large liver tumor. Blood glucose and serum calcium were greatly elevated.<\/jats:p><\/jats:sec><jats:sec><jats:title>Design and objective<\/jats:title><jats:p>Case report describing the causative relationship of the clinical findings.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Physical examination; ultrasound of the abdomen; CT scan of the abdomen and the chest; conventional X-rays; routine hematology; blood chemistry and multiple parameters of calcium and phosphorus metabolism; multisteroid analysis in serum and urine; adrenocortical stimulation and suppression tests; histopathological assessment of the resected tumor; immunohistochemistry for ACTH, \u03b2-endorphin, corticotrophin-releasing hormone (CRH), and PTH-related peptide (PTHrP); electron microscopy of tumor cells; ACTH and CRH extraction from the tumor tissue; and clinical follow-up for more than 20 years.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Giant hepatoblastoma (HB; \u223c1000\u200aml volume) of the right lobe of the liver with combined ectopic ACTH syndrome and PTHrP-induced tumor-associated hypercalcemia. Wide local excision and polychemotherapy led to complete reversal of the paraneoplastic phenotype.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>This is the first report of an endocrine-active HB causing both Cushing's syndrome and PTHrP-related \u2018humoral hypercalcemia of malignancy\u2019. This information should be added to the well-known \u03b2-human chorionic gonadotropin-related paraneoplastic effects of HB in children.<\/jats:p><\/jats:sec>","DOI":"10.1530\/eje-09-0961","type":"journal-article","created":{"date-parts":[[2010,2,5]],"date-time":"2010-02-05T02:51:56Z","timestamp":1265338316000},"page":"813-818","update-policy":"https:\/\/doi.org\/10.1530\/crossmarkpolicy-1","source":"Crossref","is-referenced-by-count":15,"title":["First report of ectopic ACTH syndrome and PTHrP-induced hypercalcemia due to a hepatoblastoma in a child"],"prefix":"10.1093","volume":"162","author":[{"given":"Thomas G P","family":"Grunewald","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Irene","family":"von Luettichau","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ulrich","family":"Welsch","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Helmuth-G\u00fcnther","family":"D\u00f6rr","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frank","family":"H\u00f6pner","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kalman","family":"Kovacs","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stefan","family":"Burdach","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wolfgang","family":"Rabl","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","reference":[{"issue":"4","key":"1_23492000","doi-asserted-by":"crossref","first-page":"185","DOI":"10.1080\/09674845.2006.11732741","volume":"63","author":"Thirkill","year":"2006","journal-title":"British journal of biomedical science","ISSN":"https:\/\/id.crossref.org\/issn\/0967-4845","issn-type":"print"},{"issue":"4","key":"2_18008231","doi-asserted-by":"publisher","first-page":"303","DOI":"10.1385\/EP:14:4:303","volume":"14","author":"DeLellis","year":"2003","journal-title":"Endocrine pathology","ISSN":"https:\/\/id.crossref.org\/issn\/1046-3976","issn-type":"print"},{"issue":"7","key":"3_13271825","doi-asserted-by":"publisher","first-page":"734","DOI":"10.1136\/jcp.37.7.734","volume":"37","author":"Beach","year":"1984","journal-title":"Journal of Clinical Pathology","ISSN":"https:\/\/id.crossref.org\/issn\/0021-9746","issn-type":"print"},{"issue":"4","key":"4_9156472","doi-asserted-by":"publisher","first-page":"331","DOI":"10.3109\/08880018909034304","volume":"6","author":"Perilongo","year":"1989","journal-title":"Pediatric hematology and oncology","ISSN":"https:\/\/id.crossref.org\/issn\/0888-0018","issn-type":"print"},{"issue":"8","key":"5_19316672","doi-asserted-by":"publisher","first-page":"4955","DOI":"10.1210\/jc.2004-2527","volume":"90","year":"2005","journal-title":"Journal of Clinical Endocrinology &amp; 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