{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,28]],"date-time":"2026-09-28T22:45:54Z","timestamp":1790635554086,"version":"4.1.0"},"reference-count":56,"publisher":"Oxford University Press (OUP)","issue":"4","license":[{"start":{"date-parts":[[2023,10,14]],"date-time":"2023-10-14T00:00:00Z","timestamp":1697241600000},"content-version":"vor","delay-in-days":13,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"I-CAH Registry project"},{"DOI":"10.13039\/501100000265","name":"Medical Research Council","doi-asserted-by":"publisher","award":["G1100236"],"award-info":[{"award-number":["G1100236"]}],"id":[{"id":"10.13039\/501100000265","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Seventh European Union Framework Program","award":["201444"],"award-info":[{"award-number":["201444"]}]},{"name":"European Society for Paediatric Endocrinology Research Unit"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,10,17]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Importance<\/jats:title>\n                  <jats:p>Testicular adrenal rest tumors (TARTs), often found in male patients with congenital adrenal hyperplasia (CAH), are benign lesions causing testicular damage and infertility. We hypothesize that chronically elevated adrenocorticotropic hormone exposure during early life may promote TART development.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Objective<\/jats:title>\n                  <jats:p>This study aimed to examine the association between commencing adequate glucocorticoid treatment early after birth and TART development.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Design and participants<\/jats:title>\n                  <jats:p>This retrospective multicenter (n = 22) open cohort study collected longitudinal clinical and biochemical data of the first 4 years of life using the I-CAH registry and included 188 male patients (median age 13 years; interquartile range: 10-17) with 21-hydroxylase deficiency (n = 181) or 11-hydroxylase deficiency (n = 7). All patients underwent at least 1 testicular ultrasound.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>TART was detected in 72 (38%) of the patients. Prevalence varied between centers. When adjusted for CAH phenotype, a delayed CAH diagnosis of &amp;gt;1 year, compared with a diagnosis within 1 month of life, was associated with a 2.6 times higher risk of TART diagnosis. TART onset was not predicted by biochemical disease control or bone age advancement in the first 4 years of life, but increased height standard deviation scores at the end of the 4-year study period were associated with a 27% higher risk of TART diagnosis.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusions and relevance<\/jats:title>\n                  <jats:p>A delayed CAH diagnosis of &amp;gt;1 year vs CAH diagnosis within 1 month after birth was associated with a higher risk of TART development, which may be attributed to poor disease control in early life.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1093\/ejendo\/lvad143","type":"journal-article","created":{"date-parts":[[2023,10,14]],"date-time":"2023-10-14T15:41:25Z","timestamp":1697298085000},"page":"460-468","source":"Crossref","is-referenced-by-count":12,"title":["Hormonal control during infancy and testicular adrenal rest tumor development in males with congenital adrenal hyperplasia: a retrospective multicenter cohort study"],"prefix":"10.1093","volume":"189","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2139-9076","authenticated-orcid":false,"given":"Mariska A M","family":"Schr\u00f6der","sequence":"first","affiliation":[{"name":"Department of Pediatrics, Amalia Children's Hospital, Radboud University Medical Center , Nijmegen , The Netherlands"},{"name":"Department of Laboratory Medicine, Radboudumc Graduate School, Radboud University Medical Center , Nijmegen , The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mihaela","family":"Neac\u015fu","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine, Radboudumc Graduate School, Radboud University Medical Center , Nijmegen , The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1439-236X","authenticated-orcid":false,"given":"Bas P H","family":"Adriaansen","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Amalia Children's Hospital, Radboud University Medical Center , Nijmegen , The Netherlands"},{"name":"Department of Laboratory Medicine, Radboudumc Graduate School, Radboud University Medical Center , Nijmegen , The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fred C G J","family":"Sweep","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine, Radboudumc Graduate School, Radboud University Medical Center , Nijmegen , The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"S Faisal","family":"Ahmed","sequence":"additional","affiliation":[{"name":"Developmental Endocrinology Research Group, University of Glasgow , Glasgow , United Kingdom"},{"name":"Office of Rare Conditions, University of Glasgow , Glasgow , United 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Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Niels Holtum","family":"Birkeb\u00e6k","sequence":"additional","affiliation":[{"name":"Department of Pediatrics and Steno Diabetes Center Aarhus, Aarhus University Hospital , Aarhus , Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christiaan","family":"de Bruin","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Leiden University Medical Center , Leiden , The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Walter","family":"Bonfig","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Technical University Munich , Munich , Germany"},{"name":"Department of Pediatrics, Klinikum Wels-Grieskirchen , Wels , Austria"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jillian","family":"Bryce","sequence":"additional","affiliation":[{"name":"Office of Rare Conditions, University of Glasgow , Glasgow , United 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Surgery, Endocrine Organs and Tissue Transplantation, MOH of Ukraine , Kyiv , Ukraine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2658-6866","authenticated-orcid":false,"given":"Tulay","family":"Guran","sequence":"additional","affiliation":[{"name":"Pediatric Endocrinology and Diabetes, Marmara University , Istanbul , Turkey"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ayla","family":"G\u00fcven","sequence":"additional","affiliation":[{"name":"Baskent University Medical Faculty, Istanbul Hospital, Pediatric Endocrinology , Istanbul , Turkey"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nermine Hussein","family":"Amr","sequence":"additional","affiliation":[{"name":"Pediatrics Department, Ain Shams University , Cairo , Egypt"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dominika","family":"Janus","sequence":"additional","affiliation":[{"name":"Department of Pediatric and 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Rita","family":"Stancampiano","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Endocrine Unit, IRCCS San Raffaele Scientific Institute, Endo-ERN Center for Rare Endocrine Conditions , Milan , Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ana","family":"Vieites","sequence":"additional","affiliation":[{"name":"Centro de Investigaciones Endocrinol\u00f3gicas Buenos Aires , Buenos Aires , Argentina"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Liat","family":"de Vries","sequence":"additional","affiliation":[{"name":"The Jesse Z and Sara Lea Shafer Institute for Endocrinology and Diabetes, National Center for Childhood Diabetes, Schneider Children's Medical Center of Israel and Felsenstein Medical Research Center at Petach Tikva, Sackler Faculty of Medicine, Tel Aviv University , Tel Aviv , Israel"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zehra","family":"Yavas 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