{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,24]],"date-time":"2026-01-24T02:08:49Z","timestamp":1769220529483,"version":"3.49.0"},"reference-count":12,"publisher":"Walter de Gruyter GmbH","issue":"4","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2019,4,24]]},"abstract":"<jats:title>Abstract<\/jats:title>\n<jats:sec id=\"j_jpem-2018-0419_s_999\">\n<jats:title id=\"d4355650e33412\">Context<\/jats:title>\n<jats:p>Germ cell tumours (GCTs) secreting \u03b2-human chorionic gonadotropin (\u03b2-HCG) are a rare cause of gonadotropin-independent precocious puberty (GIPP).<\/jats:p><\/jats:sec>\n<jats:sec id=\"j_jpem-2018-0419_s_998\">\n<jats:title id=\"d4355650e33420\">Case description<\/jats:title>\n<jats:p>A 5.7-year-old boy presented with GIPP. Investigations to elucidate the underlying cause revealed elevated serum \u03b2-HCG. Ultrasound of the abdomen and testes, urine steroid profile, bone isotope scan, and sequencing of the luteinizing hormone receptor gene (<jats:italic>LHCGR<\/jats:italic>) were normal. Despite paired serum and cerebrospinal fluid \u03b2-HCG measurement suggesting local (brain) \u03b2-HCG production, repeated magnetic resonance imaging (MRI) of the brain as well as MRI of the mediastinum did not identify a tumour source of persistently elevated serum \u03b2-HCG. Treatment with cyproterone acetate and spironolactone was unsuccessful. Increase in testicular volumes prompted the addition of a gonadotropin releasing hormone (GnRH) analogue. Due to progressing virilisation and skeletal maturation, treatment was changed to a combination of anastrozole and bicalutamide at the age of 7 years. One year later, serum \u03b2-HCG and testosterone concentrations spontaneously normalised followed by reductions in the height velocity, skeletal maturation and virilisation. The proband achieved his genetic height potential. No medication side effects were observed. The patient subsequently presented with non-secreting pineal GCT at 14 years, 8\u00bd years after his initial presentation with GIPP.<\/jats:p><\/jats:sec>\n<jats:sec id=\"j_jpem-2018-0419_s_997\">\n<jats:title id=\"d4355650e33431\">Conclusions<\/jats:title>\n<jats:p>Our case highlights that GIPP with no definite underlying aetiology at diagnosis should be considered as a prodrome for GCTs, and regular radiological surveillance for earlier tumour identification is warranted. To the best of our knowledge, our case is the first reported case of the use of anastrozole and bicalutamide in the setting of idiopathic GIPP. The good height outcome in our case warrants the trial of anastrozole and bicalutamide in similar cases.<\/jats:p><\/jats:sec>","DOI":"10.1515\/jpem-2018-0419","type":"journal-article","created":{"date-parts":[[2019,4,7]],"date-time":"2019-04-07T02:49:38Z","timestamp":1554605378000},"page":"403-407","source":"Crossref","is-referenced-by-count":6,"title":["Idiopathic gonadotropin-independent precocious puberty \u2013 is regular surveillance required?"],"prefix":"10.1515","volume":"32","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7416-003X","authenticated-orcid":false,"given":"Ved Bhushan","family":"Arya","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Justin H.","family":"Davies","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"374","reference":[{"key":"ref11","doi-asserted-by":"crossref","first-page":"1980","DOI":"10.1210\/jc.2015-3500","article-title":"The etiology and clinical features of non-CAH gonadotropin-independent precocious puberty: a multicenter study","volume":"101","year":"2016","journal-title":"J Clin Endocrinol Metab"},{"key":"ref41","doi-asserted-by":"crossref","first-page":"1264","DOI":"10.1210\/jc.2013-3724","article-title":"Central diabetes insipidus in children and young adults: etiological diagnosis and long-term outcome of idiopathic cases","volume":"99","year":"2014","journal-title":"J Clin Endocrinol Metab"},{"key":"ref61","doi-asserted-by":"crossref","first-page":"94","DOI":"10.1007\/s12149-013-0787-4","article-title":"Clinical use of (11)C-methionine and (18)F-FDG-PET for germinoma in central nervous system","volume":"28","year":"2014","journal-title":"Ann Nucl Med"},{"key":"ref31","doi-asserted-by":"crossref","first-page":"183","DOI":"10.6065\/apem.2013.18.4.183","article-title":"Boys with precocious or early puberty: incidence of pathological brain magnetic resonance imaging findings and factors related to newly developed brain lesions","volume":"18","year":"2013","journal-title":"Ann Pediatr Endocrinol Metab"},{"key":"ref51","doi-asserted-by":"crossref","first-page":"1668","DOI":"10.1212\/01.wnl.0000261908.36803.ac","article-title":"CNS germ cell tumor (CNSGCT) of childhood: presentation and delayed diagnosis","volume":"68","year":"2007","journal-title":"Neurology"},{"key":"ref111","first-page":"999","article-title":"Bicalutamide plus anastrozole for the treatment of gonadotropin-independent precocious puberty in boys with testotoxicosis: a phase II, open-label pilot study (BATT)","volume":"23","year":"2010","journal-title":"J Pediatr Endocrinol Metab"},{"key":"ref71","first-page":"1195","article-title":"hCG-secreting pineal teratoma causing precocious puberty: report of two patients and review of the literature","volume":"15","year":"2002","journal-title":"J Pediatr Endocrinol Metab"},{"key":"ref81","doi-asserted-by":"crossref","first-page":"34","DOI":"10.1016\/j.gore.2017.06.008","article-title":"The \u201chook effect\u201d causing a negative pregnancy test in a patient with an advanced molar pregnancy","volume":"21","year":"2017","journal-title":"Gynecol Oncol Rep"},{"key":"ref01","doi-asserted-by":"crossref","first-page":"690","DOI":"10.1634\/theoncologist.2008-0037","article-title":"Pediatric central nervous system germ cell tumors: a review","volume":"13","year":"2008","journal-title":"Oncologist"},{"key":"ref21","doi-asserted-by":"crossref","first-page":"786","DOI":"10.1111\/cen.12416","article-title":"Mixed germ cell tumour after testotoxicosis","volume":"81","year":"2014","journal-title":"Clin Endocrinol (Oxf)"},{"key":"ref101","first-page":"135","article-title":"Aromatase inhibitors in pediatrics","volume":"8","year":"2011","journal-title":"Nat Rev Endocrinol"},{"key":"ref91","doi-asserted-by":"crossref","first-page":"416","DOI":"10.1016\/j.jpeds.2006.04.027","article-title":"Treatment of familial male-limited precocious puberty with bicalutamide and anastrozole","volume":"149","year":"2006","journal-title":"J Pediatr"}],"container-title":["Journal of Pediatric Endocrinology and Metabolism"],"original-title":[],"link":[{"URL":"http:\/\/www.degruyter.com\/view\/j\/jpem.2019.32.issue-4\/jpem-2018-0419\/jpem-2018-0419.xml","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/www.degruyter.com\/document\/doi\/10.1515\/jpem-2018-0419\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,4,22]],"date-time":"2021-04-22T01:44:51Z","timestamp":1619055891000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.degruyter.com\/document\/doi\/10.1515\/jpem-2018-0419\/html"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2019,4,24]]},"references-count":12,"journal-issue":{"issue":"4"},"URL":"https:\/\/doi.org\/10.1515\/jpem-2018-0419","relation":{},"ISSN":["2191-0251","0334-018X"],"issn-type":[{"value":"2191-0251","type":"electronic"},{"value":"0334-018X","type":"print"}],"subject":[],"published":{"date-parts":[[2019,4,24]]}}}