{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,5,9]],"date-time":"2025-05-09T15:36:11Z","timestamp":1746804971343,"version":"3.40.5"},"reference-count":16,"publisher":"Wiley","license":[{"start":{"date-parts":[[2016,1,1]],"date-time":"2016-01-01T00:00:00Z","timestamp":1451606400000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Case Reports in Endocrinology"],"published-print":{"date-parts":[[2016]]},"abstract":"<jats:p>Pegvisomant (PEGv) is a growth hormone receptor antagonist approved for the treatment of acromegaly; one of its documented adverse effects is reversible elevation of hepatic enzymes. We report a 39-year-old male acromegalic patient with a pituitary macroadenoma who underwent transsphenoidal surgery. The patient\u2019s condition improved but GH and IGF-I levels did not normalize; as a consequence, we first administered dopamine agonists and then somatostatin receptor ligands (SRLs) with poor response. PEGv 15\u2009mg every other day was added to lanreotide 120\u2009mg monthly. The patient developed a severe hepatitis five months after starting the combination therapy. Elevated ferritin, iron, and transferrin saturation suggested probable hepatitis due to haemochromatosis. We performed a liver biopsy which showed an acute cholestatic hepatitis consistent with toxic etiology. A heterozygous genotype<jats:italic>UGT1A1<\/jats:italic><mml:math xmlns:mml=\"http:\/\/www.w3.org\/1998\/Math\/MathML\" id=\"M2\"><mml:mrow><mml:msup><mml:mrow><mml:mi>\u200b<\/mml:mi><\/mml:mrow><mml:mrow><mml:mo mathvariant=\"bold\">\u204e<\/mml:mo><mml:mi mathvariant=\"normal\">\ue899<\/mml:mi><\/mml:mrow><\/mml:msup><\/mml:mrow><\/mml:math><jats:italic>28<\/jats:italic>polymorphism associated with Gilbert\u2019s syndrome was also found in this Argentine patient. The predominant clinical presentation resembled an acute cholestatic hepatitis associated with severe hemosiderosis, a different and new pattern of PEGv hepatotoxicity.<\/jats:p>","DOI":"10.1155\/2016\/2087102","type":"journal-article","created":{"date-parts":[[2016,2,9]],"date-time":"2016-02-09T21:01:23Z","timestamp":1455051683000},"page":"1-5","source":"Crossref","is-referenced-by-count":1,"title":["Pegvisomant-Induced Cholestatic Hepatitis in an Acromegalic Patient with UGT1A1<mml:math xmlns:mml=\"http:\/\/www.w3.org\/1998\/Math\/MathML\" id=\"M1\"><mml:mo>\u200a<\/mml:mo><mml:msup><mml:mrow><mml:mi>\u200b<\/mml:mi><\/mml:mrow><mml:mrow><mml:mi mathvariant=\"normal\">\ue899<\/mml:mi><mml:mo mathvariant=\"bold\">\u204e<\/mml:mo><mml:mi mathvariant=\"normal\">\ue899<\/mml:mi><\/mml:mrow><\/mml:msup><mml:mo>\u200a<\/mml:mo><\/mml:math>28 Mutation"],"prefix":"10.1155","volume":"2016","author":[{"given":"Maria Susana","family":"Mallea-Gil","sequence":"first","affiliation":[{"name":"Servicio de Endocrinolog\u0131a, Hospital Militar Central, 726 Luis Mar\u00eda Campos Avenue, 1425 Buenos Aires, Argentina"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ignacio","family":"Bernabeu","sequence":"additional","affiliation":[{"name":"Endocrinology Division and Fundacion Publica Galega de Medicina Xenomica (Unidad de Medicina Molecular), Complejo Hospitalario Universitario de Santiago de Compostela, Universidad de Santiago de Compostela, Travesia Choupana s\/n, Santiago de Compostela, 15706 La Coru\u00f1a, Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Adriana","family":"Spiraquis","sequence":"additional","affiliation":[{"name":"Servicio de Gastroenterolog\u0131a, Hospital Militar Central, 726 Luis Mar\u00eda Campos Avenue, 1425 Buenos Aires, Argentina"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Alejandra","family":"Avangina","sequence":"additional","affiliation":[{"name":"Departamento de Anatom\u0131a Patologica, Hospital de Cl\u00ednicas, Universidad de Buenos Aires, 2351 C\u00f3rdoba Avenue, 1120 Buenos Aires, Argentina"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lourdes","family":"Loidi","sequence":"additional","affiliation":[{"name":"Endocrinology Division and Fundacion Publica Galega de Medicina Xenomica (Unidad de Medicina Molecular), Complejo Hospitalario Universitario de Santiago de Compostela, Universidad de Santiago de Compostela, Travesia Choupana s\/n, Santiago de Compostela, 15706 La Coru\u00f1a, Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Carolina","family":"Ballarino","sequence":"additional","affiliation":[{"name":"Servicio de Endocrinolog\u0131a, Hospital Militar Central, 726 Luis Mar\u00eda Campos Avenue, 1425 Buenos Aires, Argentina"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","reference":[{"key":"1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMra062453"},{"key":"2","doi-asserted-by":"publisher","DOI":"10.1016\/j.ecl.2007.10.002"},{"key":"3","doi-asserted-by":"publisher","DOI":"10.1530\/eje.1.02079"},{"key":"4","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2009-2547"},{"key":"5","doi-asserted-by":"publisher","DOI":"10.1530\/eje.1.02312"},{"key":"6","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2011-2508"},{"key":"17","doi-asserted-by":"publisher","DOI":"10.1530\/eje-09-0040"},{"key":"8","doi-asserted-by":"publisher","DOI":"10.1016\/0168-8278(90)90124-a"},{"key":"9","doi-asserted-by":"publisher","DOI":"10.1002\/hep.24229"},{"key":"10","doi-asserted-by":"publisher","DOI":"10.1016\/j.mayocp.2013.09.016"},{"issue":"38, supplement 2","key":"11","first-page":"s44","volume":"1","year":"2004","journal-title":"Clinical Infectious Diseases"},{"key":"12","doi-asserted-by":"publisher","DOI":"10.1136\/gut.2008.163675"},{"issue":"3","key":"13","doi-asserted-by":"crossref","first-page":"401","DOI":"10.1111\/j.1365-2265.1994.tb03938.x","volume":"40","year":"1994","journal-title":"Clinical Endocrinology"},{"issue":"7","key":"14","doi-asserted-by":"crossref","first-page":"467","DOI":"10.1155\/2006\/357340","volume":"20","year":"2006","journal-title":"Canadian Journal of Gastroenterology"},{"key":"15","doi-asserted-by":"publisher","DOI":"10.1530\/EJE-13-0657"},{"key":"16","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2014-2032"}],"container-title":["Case Reports in Endocrinology"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/downloads.hindawi.com\/journals\/crie\/2016\/2087102.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/downloads.hindawi.com\/journals\/crie\/2016\/2087102.xml","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/downloads.hindawi.com\/journals\/crie\/2016\/2087102.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,6,14]],"date-time":"2024-06-14T08:07:19Z","timestamp":1718352439000},"score":1,"resource":{"primary":{"URL":"http:\/\/www.hindawi.com\/journals\/crie\/2016\/2087102\/"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2016]]},"references-count":16,"alternative-id":["2087102","2087102"],"URL":"https:\/\/doi.org\/10.1155\/2016\/2087102","relation":{},"ISSN":["2090-6501","2090-651X"],"issn-type":[{"type":"print","value":"2090-6501"},{"type":"electronic","value":"2090-651X"}],"subject":[],"published":{"date-parts":[[2016]]}}}