{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,10]],"date-time":"2026-07-10T15:58:40Z","timestamp":1783699120969,"version":"3.55.0"},"reference-count":37,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2019,5,7]],"date-time":"2019-05-07T00:00:00Z","timestamp":1557187200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Clinical Endocrinology"],"published-print":{"date-parts":[[2019,8]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Objective<\/jats:title><jats:p>Adrenonodular hyperplasia and tumour formation are potential long\u2010term complications of congenital adrenal hyperplasia (CAH) with little known regarding the clinical implications. Our aim was to describe volumetric adrenal morphology and determine the association between radiological findings and comorbidities in adults with classic CAH.<\/jats:p><\/jats:sec><jats:sec><jats:title>Design<\/jats:title><jats:p>This was a cross\u2010sectional study of 88 patients (mean age 29.2\u00a0\u00b1\u00a013\u00a0years, 47 females) with classic CAH seen in a tertiary referral centre.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>CT imaging, performed at study entry or when reaching adulthood, was used to create 3\u2010dimensional volumetric models. Clinical, genetic and hormonal evaluations were collected and correlated with adrenal morphology and tumour formation.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Over one\u2010third of the cohort was obese. 53% had elevated 17\u2010OH\u2010progesterone or androstenedione; and 60% had adrenal hyperplasia. Tumours included 11 myelolipomas, 8 benign adrenocortical\u00a0adenomas, 1 pheochromocytoma and 50% of men had testicular adrenal rest tissue. CAH patients with adrenal hyperplasia had significantly higher number of comorbidities than those with morphologically\u00a0normal adrenals (<jats:italic>P<\/jats:italic>\u00a0=\u00a00.03). Variables that positively correlated with adrenal volume included hypogonadal\/oligomenorrhoeic status, hypertension, androstenedione, aldosterone, and triglyceride levels, and in women, low HDL and insulin resistance. Elevated aldosterone was observed in a subset of patients with simple virilizing CAH.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Adrenocortical hyperplasia is associated with a number of comorbidities, especially hypogonadism. Aldosterone production associated with adrenal enlargement may play a role in the development of metabolic risk factors. Further studies are needed to assess the long\u2010term impact of the excess adrenal steroid milieu associated with adrenal enlargement to develop improved management strategies for CAH.<\/jats:p><\/jats:sec>","DOI":"10.1111\/cen.13996","type":"journal-article","created":{"date-parts":[[2019,4,19]],"date-time":"2019-04-19T02:34:48Z","timestamp":1555641288000},"page":"247-255","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":19,"title":["Adrenal morphology and associated comorbidities in congenital adrenal hyperplasia"],"prefix":"10.1111","volume":"91","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0936-2249","authenticated-orcid":false,"given":"Diala","family":"El\u2010Maouche","sequence":"first","affiliation":[{"name":"The National Institutes of Health Clinical Center Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fady","family":"Hannah\u2010Shmouni","sequence":"additional","affiliation":[{"name":"The <i>Eunice Kennedy Shriver<\/i> National Institute of Child Health and Human Development Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0637-4559","authenticated-orcid":false,"given":"Ashwini","family":"Mallappa","sequence":"additional","affiliation":[{"name":"The National Institutes of Health Clinical Center Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Courtney J.","family":"Hargreaves","sequence":"additional","affiliation":[{"name":"The National Institutes of Health Clinical Center Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nilo A.","family":"Avila","sequence":"additional","affiliation":[{"name":"National Heart, Lung and Blood Institute Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3746-0460","authenticated-orcid":false,"given":"Deborah P.","family":"Merke","sequence":"additional","affiliation":[{"name":"The National Institutes of Health Clinical Center Bethesda Maryland"},{"name":"The <i>Eunice Kennedy Shriver<\/i> National Institute of Child Health and Human Development Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2019,5,7]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2018-01865"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(17)31431-9"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2008-2380"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12020-013-0162-1"},{"issue":"3","key":"e_1_2_9_6_1","first-page":"685","article-title":"Adrenal incidentaloma and patients with homozygous or heterozygous congenital adrenal hyperplasia","volume":"74","author":"Jaresch S","year":"1992","journal-title":"J Clin Endocrinol Metab"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2265.2011.04151.x"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2009-1929"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2012-2102"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2010-0917"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1210\/JC.2015-2093"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2013-1440"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2016-3989"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1001\/jama.285.19.2486"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2009-2616"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2016-1916"},{"key":"e_1_2_9_17_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2018-00241"},{"key":"e_1_2_9_18_1","doi-asserted-by":"publisher","DOI":"10.2337\/diacare.26.12.3320"},{"key":"e_1_2_9_19_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2018-00229"},{"key":"e_1_2_9_20_1","doi-asserted-by":"publisher","DOI":"10.1055\/s-0042-103686"},{"key":"e_1_2_9_21_1","doi-asserted-by":"publisher","DOI":"10.1055\/s-0043-107245"},{"key":"e_1_2_9_22_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00261-003-0139-2"},{"key":"e_1_2_9_23_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.acra.2014.06.008"},{"key":"e_1_2_9_24_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00330-015-4063-y"},{"key":"e_1_2_9_25_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00198-014-2794-2"},{"key":"e_1_2_9_26_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2010-0319"},{"key":"e_1_2_9_27_1","doi-asserted-by":"publisher","DOI":"10.1007\/978-3-319-44136-8_11"},{"key":"e_1_2_9_28_1","doi-asserted-by":"publisher","DOI":"10.1210\/en.2016-1804"},{"key":"e_1_2_9_29_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2265.2010.03928.x"},{"key":"e_1_2_9_30_1","unstructured":"AdultC.Obesity Prevalence Maps.https:\/\/www.cdc.gov\/obesity\/data\/prevalence-maps.html. Accessed January 16 2019."},{"key":"e_1_2_9_31_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1536-1632(02)00018-5"},{"key":"e_1_2_9_32_1","doi-asserted-by":"publisher","DOI":"10.1007\/s002610000061"},{"key":"e_1_2_9_33_1","first-page":"180","article-title":"Concerning the function and origin of the reticular zone of the adrenal corrtex; hyperplasia in the adrenogenital syndrome","volume":"78","author":"Ss BJ","year":"1946","journal-title":"Bull Johns Hopkins Hosp"},{"key":"e_1_2_9_34_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM200011093431903"},{"key":"e_1_2_9_35_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajpath.2012.05.026"},{"key":"e_1_2_9_36_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2018-00217"},{"key":"e_1_2_9_37_1","doi-asserted-by":"publisher","DOI":"10.1530\/EJE-11-0828"},{"key":"e_1_2_9_38_1","doi-asserted-by":"publisher","DOI":"10.1111\/cen.12033"}],"container-title":["Clinical Endocrinology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fcen.13996","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/cen.13996","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/cen.13996","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/cen.13996","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,10]],"date-time":"2023-09-10T06:15:57Z","timestamp":1694326557000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/cen.13996"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2019,5,7]]},"references-count":37,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2019,8]]}},"alternative-id":["10.1111\/cen.13996"],"URL":"https:\/\/doi.org\/10.1111\/cen.13996","archive":["Portico"],"relation":{},"ISSN":["0300-0664","1365-2265"],"issn-type":[{"value":"0300-0664","type":"print"},{"value":"1365-2265","type":"electronic"}],"subject":[],"published":{"date-parts":[[2019,5,7]]},"assertion":[{"value":"2018-11-04","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2019-04-15","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2019-05-07","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}