{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,29]],"date-time":"2026-07-29T19:36:04Z","timestamp":1785353764798,"version":"3.55.0"},"reference-count":38,"publisher":"Walter de Gruyter GmbH","issue":"8","funder":[{"name":"Faculty of Medicine Grant, Diponegoro University","award":["751\/UN7.F4\/PP\/IV\/2023"],"award-info":[{"award-number":["751\/UN7.F4\/PP\/IV\/2023"]}]},{"name":"European Society for Paediatric Endocrinology (ESPE) sponsored program \u201cESPE Visiting Professorship\u201d supported by Pfizer"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,8,26]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec id=\"j_jpem-2025-0026_abs_001\">\n                  <jats:title>Objectives<\/jats:title>\n                  <jats:p>Male patients with congenital adrenal hyperplasia (CAH) due to 21-hydroxylase deficiency (21OHD) are at risk of developing testicular adrenal rest tumors (TART), which are benign but an important cause of infertility. The limited medical infrastructure and lack of knowledge and awareness of 21OHD in Indonesia has contributed to missed or delayed diagnoses, leading to a reportedly low prevalence of 21OHD and an expected high rate of undiagnosed patients. In Indonesia, TART has not been widely reported, with only a single patient documented in a recent paper. This study aim was to define the potential risk factors for developing TART in Indonesian pediatric patients with 21OHD.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_jpem-2025-0026_abs_002\">\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>This cross-sectional study was conducted in 20 boys with genetically proven 21OHD (0\u201318\u00a0years old). All had pathogenic variants associated with 0\u202f% or 0\u20131% residual enzymatic activity. Medical history, clinical characteristics, genetic analysis results, biochemical measurements, bone age, and scrotal ultrasonography (US) data were evaluated.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_jpem-2025-0026_abs_003\">\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>In 5 of 20 patients (25\u202f%), TART was detected by US. The patients with TART were mainly in puberty (p=0.014). Higher androstenedione levels and advanced bone age were associated with\u00a0TART.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_jpem-2025-0026_abs_004\">\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>TART is an important complication in Indonesian patients with 21OHD presenting before or during puberty. Factors associated with TART are pubertal age and longstanding poor hormonal control. Early diagnosis and optimization of treatment may help to prevent TART development and consequently improve fertility in these patients.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1515\/jpem-2025-0026","type":"journal-article","created":{"date-parts":[[2025,7,10]],"date-time":"2025-07-10T08:02:04Z","timestamp":1752134524000},"page":"838-847","source":"Crossref","is-referenced-by-count":1,"title":["Testicular adrenal rest tumors in Indonesian boys with congenital adrenal hyperplasia"],"prefix":"10.1515","volume":"38","author":[{"ORCID":"https:\/\/orcid.org\/0009-0000-8691-8979","authenticated-orcid":false,"given":"Epifani A.","family":"Chandra","sequence":"first","affiliation":[{"name":"Faculty of Medicine , Diponegoro University , Semarang , Indonesia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5965-1981","authenticated-orcid":false,"given":"Agustini","family":"Utari","sequence":"additional","affiliation":[{"name":"Division of Pediatric Endocrinology, Department of Pediatrics, Faculty of Medicine , Diponegoro University , Semarang , Indonesia"},{"name":"Center for Biomedical Research (CEBIOR), Faculty of Medicine , Diponegoro University , Semarang , Indonesia"}],"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":"Division of Pediatric Endocrinology, Department of Pediatrics , Amalia Children\u2019s Hospital, Radboud University Medical Centre , Nijmegen , the Netherlands"},{"name":"Department of Laboratory Medicine , Radboud University Medical Centre , Nijmegen , the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5567-1704","authenticated-orcid":false,"given":"Dineke","family":"Westra","sequence":"additional","affiliation":[{"name":"Department of Human Genetics , Radboud University Medical Center , Nijmegen , the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-8373-5287","authenticated-orcid":false,"given":"Farah H.","family":"Ningrum","sequence":"additional","affiliation":[{"name":"Department of Radiology, Faculty of Medicine , Diponegoro University , Semarang , Indonesia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2600-7617","authenticated-orcid":false,"given":"Antonius G.","family":"Santoso","sequence":"additional","affiliation":[{"name":"Department of Radiology, Faculty of Medicine , Diponegoro University , Semarang , Indonesia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5417-8272","authenticated-orcid":false,"given":"Antonius E.","family":"van Herwaarden","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine , Radboud University Medical Centre , Nijmegen , the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0181-0403","authenticated-orcid":false,"given":"Hedi L.","family":"Claahsen-van der Grinten","sequence":"additional","affiliation":[{"name":"Division of Pediatric Endocrinology, Department of Pediatrics , Amalia Children\u2019s Hospital, Radboud University Medical Centre , Nijmegen , the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"374","published-online":{"date-parts":[[2025,6,20]]},"reference":[{"key":"2025080412565947875_j_jpem-2025-0026_ref_001","doi-asserted-by":"crossref","unstructured":"Claahsen-van der Grinten, HL, Speiser, PW, Ahmed, SF, Arlt, W, Auchus, RJ, Falhammar, H, et al.. 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