{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,20]],"date-time":"2026-07-20T14:17:30Z","timestamp":1784557050995,"version":"3.55.0"},"reference-count":30,"publisher":"Bioscientifica","issue":"8","license":[{"start":{"date-parts":[[2023,5,31]],"date-time":"2023-05-31T00:00:00Z","timestamp":1685491200000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["ec.bioscientifica.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2023,7,14]]},"abstract":"<jats:p>Congenital adrenal hyperplasia (CAH) is a recessive condition that affects the adrenal glands. Despite life-long replacement therapy with glucocorticoids and mineralocorticoids, adult patients with CAH often experience impaired gonadal function. In pubertal boys and in men with CAH, circulating testosterone is produced by the adrenal glands as well as the testicular, steroidogenic cells. In this European two-center study, we evaluated the function of Leydig and Sertoli cells in 61 boys and men with CAH, primarily due to 21-hydroxylase deficiency. Despite conventional hormone replacement therapy, our results indicated a significant reduction in serum concentrations of both Leydig cell-derived hormones (i.e. insulin-like factor 3 (INSL3) and testosterone) and Sertoli cell-derived hormones (i.e. inhibin B and anti-M\u00fcllerian hormone) in adult males with CAH. Serum concentrations of INSL3 were particularly reduced in those with testicular adrenal rest tumors. To our knowledge, this is the first study to evaluate circulating INSL3 as a candidate biomarker to monitor Leydig cell function in patients with CAH.<\/jats:p>","DOI":"10.1530\/ec-23-0073","type":"journal-article","created":{"date-parts":[[2023,5,31]],"date-time":"2023-05-31T11:46:39Z","timestamp":1685533599000},"update-policy":"https:\/\/doi.org\/10.1530\/crossmarkpolicy-10","source":"Crossref","is-referenced-by-count":9,"title":["Reduced serum concentrations of biomarkers reflecting Leydig and Sertoli cell function in male patients with congenital adrenal hyperplasia"],"prefix":"10.1530","volume":"12","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-3303-5352","authenticated-orcid":true,"given":"Trine Holm","family":"Johannsen","sequence":"first","affiliation":[{"name":"Department of Growth and Reproduction, Copenhagen University Hospital - Rigshospitalet, Copenhagen, Denmark"},{"name":"International Centre for Research and Research Training in Endocrine Disruption of Male Reproduction and Child Health (EDMaRC), Rigshospitalet, University of Copenhagen, Copenhagen, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2842-1128","authenticated-orcid":true,"given":"Jakob","family":"Albrethsen","sequence":"additional","affiliation":[{"name":"Department of Growth and Reproduction, Copenhagen University Hospital - Rigshospitalet, Copenhagen, Denmark"},{"name":"International Centre for Research and Research Training in Endocrine Disruption of Male Reproduction and Child Health (EDMaRC), Rigshospitalet, University of Copenhagen, Copenhagen, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6890-8395","authenticated-orcid":true,"given":"Vassos","family":"Neocleous","sequence":"additional","affiliation":[{"name":"The Cyprus Institute of Neurology and Genetics, Department of Molecular Genetics, Function and Therapy, Nicosia, Cyprus"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Federico","family":"Baronio","sequence":"additional","affiliation":[{"name":"S. Orsola-Malpighi University Hospital, Department of Medical and Surgical Sciences, Bologna, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9552-4899","authenticated-orcid":true,"given":"Martine","family":"Cools","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Division of Pediatric Endocrinology, Ghent University Hospital and Department of Internal Medicine and Pediatrics, Ghent University, Ghent, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7885-7467","authenticated-orcid":true,"given":"Lise","family":"Aksglaede","sequence":"additional","affiliation":[{"name":"Department of Growth and Reproduction, Copenhagen University Hospital - Rigshospitalet, Copenhagen, Denmark"},{"name":"International Centre for Research and Research Training in Endocrine Disruption of Male Reproduction and Child Health (EDMaRC), Rigshospitalet, University of Copenhagen, Copenhagen, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4827-0838","authenticated-orcid":true,"given":"Niels","family":"J\u00f8rgensen","sequence":"additional","affiliation":[{"name":"Department of Growth and Reproduction, Copenhagen University Hospital - Rigshospitalet, Copenhagen, Denmark"},{"name":"International Centre for Research and Research Training in Endocrine Disruption of Male Reproduction and Child Health (EDMaRC), Rigshospitalet, University of Copenhagen, Copenhagen, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peter","family":"Christiansen","sequence":"additional","affiliation":[{"name":"Department of Growth and Reproduction, Copenhagen University Hospital - Rigshospitalet, Copenhagen, Denmark"},{"name":"International Centre for Research and Research Training in Endocrine Disruption of Male Reproduction and Child Health (EDMaRC), Rigshospitalet, University of Copenhagen, Copenhagen, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Meropi","family":"Toumba","sequence":"additional","affiliation":[{"name":"The Cyprus Institute of Neurology and Genetics, Department of Molecular Genetics, Function and Therapy, Nicosia, Cyprus"},{"name":"Pediatric Endocrinology Clinic, Department of Pediatrics, Aretaeio Hospital, Nicosia, Cyprus"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4590-4778","authenticated-orcid":true,"given":"Pavlos","family":"Fanis","sequence":"additional","affiliation":[{"name":"The Cyprus Institute of Neurology and Genetics, Department of Molecular Genetics, Function and Therapy, Nicosia, Cyprus"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7418-6878","authenticated-orcid":true,"given":"Marie Lindhardt","family":"Ljubicic","sequence":"additional","affiliation":[{"name":"Department of Growth and Reproduction, Copenhagen University Hospital - Rigshospitalet, Copenhagen, Denmark"},{"name":"International Centre for Research and Research Training in Endocrine Disruption of Male Reproduction and Child Health (EDMaRC), Rigshospitalet, University of Copenhagen, Copenhagen, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0534-4350","authenticated-orcid":true,"given":"Anders","family":"Juul","sequence":"additional","affiliation":[{"name":"Department of Growth and Reproduction, Copenhagen University Hospital - Rigshospitalet, Copenhagen, Denmark"},{"name":"International Centre for Research and Research Training in Endocrine Disruption of Male Reproduction and Child Health (EDMaRC), Rigshospitalet, University of Copenhagen, Copenhagen, Denmark"},{"name":"Department of Clinical Medicine, University of Copenhagen, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"416","reference":[{"key":"bib1","series-title":"Endocrine Reviews","first-page":"91","article-title":"Congenital adrenal hyperplasia - current insights in pathophysiology, diagnostics, and management","volume":"43","author":"Claahsen-van der Grinten","year":"2022","unstructured":"Claahsen-van der Grinten HLSpeiser PWAhmed SFArlt WAuchus RJFalhammar HFl\u00fcck CEGuasti LHuebner AKortmann BBM, et al. Congenital adrenal hyperplasia - current insights in pathophysiology, diagnostics, and management. Endocrine Reviews20224391\u2013159. (https:\/\/doi.org\/10.1210\/endrev\/bnab016)"},{"key":"bib2","series-title":"Lancet","first-page":"2125","article-title":"Congenital adrenal hyperplasia","volume":"365","author":"Merke","year":"2005","unstructured":"Merke DP & Bornstein SR. Congenital adrenal hyperplasia. Lancet20053652125\u20132136. (https:\/\/doi.org\/10.1016\/S0140-6736(0566736-0)"},{"key":"bib3","series-title":"Nature Reviews. Endocrinology","first-page":"115","article-title":"Treatment and health outcomes in adults with congenital adrenal hyperplasia","volume":"10","author":"Han","year":"2014","unstructured":"Han TSWalker BRArlt W & Ross RJ. Treatment and health outcomes in adults with congenital adrenal hyperplasia. Nature Reviews. Endocrinology201410115\u2013124. (https:\/\/doi.org\/10.1038\/nrendo.2013.239)"},{"key":"bib4","series-title":"International Journal of Andrology","first-page":"521","article-title":"Testicular adrenal rest tumours in boys, adolescents and adult men with congenital adrenal hyperplasia may be associated with the CYP21A2 mutation","volume":"33","author":"Mouritsen","year":"2010","unstructured":"Mouritsen AJ\u00f8rgensen NMain KMSchwartz M & Juul A. Testicular adrenal rest tumours in boys, adolescents and adult men with congenital adrenal hyperplasia may be associated with the CYP21A2 mutation. International Journal of Andrology201033521\u2013527. (https:\/\/doi.org\/10.1111\/j.1365-2605.2009.00967.x)"},{"key":"bib5","series-title":"Endocrine Reviews","first-page":"973","article-title":"Testicular adrenal rest tumors: current insights on prevalence, characteristics, origin, and treatment","volume":"40","author":"Engels","year":"2019","unstructured":"Engels MSpan PNHerwaarden van AESweep FCGJStikkelbroeck NMML & Claahsen-van der Grinten HL. Testicular adrenal rest tumors: current insights on prevalence, characteristics, origin, and treatment. Endocrine Reviews201940973\u2013987. (https:\/\/doi.org\/10.1210\/er.2018-00258)"},{"key":"bib6","series-title":"Journal of Clinical Endocrinology and Metabolism","first-page":"E1820","article-title":"Testicular adrenal rest tumors develop independently of long-term disease control: a longitudinal analysis of 50 adult men with congenital adrenal hyperplasia due to classic 21-hydroxylase deficiency","volume":"98","author":"Reisch","year":"2013","unstructured":"Reisch NRottenkolber MGreifenstein AKrone NSchmidt HReincke MSchwarz HP & Beuschlein F. Testicular adrenal rest tumors develop independently of long-term disease control: a longitudinal analysis of 50 adult men with congenital adrenal hyperplasia due to classic 21-hydroxylase deficiency. Journal of Clinical Endocrinology and Metabolism201398E1820\u2013E1826. (https:\/\/doi.org\/10.1210\/jc.2012-3181)"},{"key":"bib7","series-title":"Journal of Clinical Endocrinology and Metabolism","first-page":"1108","article-title":"The effects of gonadotropin suppression and selective replacement on insulin-like factor 3 secretion in normal adult men","volume":"91","author":"Bay","year":"2006","unstructured":"Bay KMatthiesson KLMcLachlan RI & Andersson AM. The effects of gonadotropin suppression and selective replacement on insulin-like factor 3 secretion in normal adult men. Journal of Clinical Endocrinology and Metabolism2006911108\u20131111. (https:\/\/doi.org\/10.1210\/jc.2005-1865)"},{"key":"bib8","series-title":"Clinical Chemistry and Laboratory Medicine","first-page":"1913","article-title":"Development and validation of a mass spectrometry-based assay for quantification of insulin-like factor 3 in human serum","volume":"56","author":"Albrethsen","year":"2018","unstructured":"Albrethsen JFrederiksen HAndersson AMAnand-Ivell RNordkap LBang AKJ\u00f8rgensen N & Juul A. Development and validation of a mass spectrometry-based assay for quantification of insulin-like factor 3 in human serum. Clinical Chemistry and Laboratory Medicine2018561913\u20131920. (https:\/\/doi.org\/10.1515\/cclm-2018-0171)"},{"key":"bib9","series-title":"Journal of Clinical Endocrinology and Metabolism","first-page":"1868","article-title":"Evaluation of serum insulin-like factor 3 quantification by LC-MS\/MS as a biomarker of Leydig cell function","volume":"105","author":"Albrethsen","year":"2020","unstructured":"Albrethsen JJohannsen THJ\u00f8rgensen NFrederiksen HSennels HPJ\u00f8rgensen HLFahrenkrug JPetersen JHLinneberg ANordkap L, et al. Evaluation of serum insulin-like factor 3 quantification by LC-MS\/MS as a biomarker of Leydig cell function. Journal of Clinical Endocrinology and Metabolism20201051868\u20131877. (https:\/\/doi.org\/10.1210\/clinem\/dgaa145)"},{"key":"bib10","series-title":"Endocrine","first-page":"578","article-title":"Serum insulin-like factor 3 quantification by LC\u2013MS\/MS in male patients with hypogonadotropic hypogonadism and Klinefelter syndrome","volume":"71","author":"Johannsen","year":"2021","unstructured":"Johannsen THLjubicic MLYoung JTrabado SPetersen JHLinneberg AAlbrethsen J & Juul A. Serum insulin-like factor 3 quantification by LC\u2013MS\/MS in male patients with hypogonadotropic hypogonadism and Klinefelter syndrome. Endocrine202171578\u2013585. (https:\/\/doi.org\/10.1007\/s12020-021-02609-0)"},{"key":"bib11","series-title":"Archives of Internal Medicine","first-page":"863","article-title":"A formula to estimate the approximate surface area if height and weight be known","volume":"17","author":"Du Bois","year":"1916","unstructured":"Du Bois D & Du Bois EF. A formula to estimate the approximate surface area if height and weight be known. Archives of Internal Medicine191617863\u2013871."},{"key":"bib12","series-title":"Allergy, Asthma, and Clinical Immunology","first-page":"30","article-title":"A practical guide to the monitoring and management of the complications of systemic corticosteroid therapy","volume":"9","author":"Liu","year":"2013","unstructured":"Liu DAhmet AWard LKrishnamoorthy PMandelcorn EDLeigh RBrown JPCohen A & Kim H. A practical guide to the monitoring and management of the complications of systemic corticosteroid therapy. Allergy, Asthma, and Clinical Immunology2013930. (https:\/\/doi.org\/10.1186\/1710-1492-9-30)"},{"key":"bib13","series-title":"Human Reproduction","first-page":"2323","article-title":"The LH\/FSH ratio is not a sex-dimorphic marker after infancy: data from 6417 healthy individuals and 125 patients with Differences of Sex Development","volume":"35","author":"Ljubicic","year":"2020","unstructured":"Ljubicic MLJespersen KAksglaede LHagen CPPetersen JHAndersen HRLinneberg AMain KMAndersson AMJohannsen TH, et al. The LH\/FSH ratio is not a sex-dimorphic marker after infancy: data from 6417 healthy individuals and 125 patients with Differences of Sex Development. Human Reproduction2020352323\u20132335. (https:\/\/doi.org\/10.1093\/humrep\/deaa182)"},{"key":"bib14","series-title":"Clinica Chimica Acta","first-page":"180","article-title":"Isotope-dilution TurboFlow-LC-MS\/MS method for simultaneous quantification of ten steroid metabolites in serum","volume":"468","author":"S\u00f8eborg","year":"2017","unstructured":"S\u00f8eborg TFrederiksen HJohannsen THAndersson AM & Juul A. Isotope-dilution TurboFlow-LC-MS\/MS method for simultaneous quantification of ten steroid metabolites in serum. Clinica Chimica Acta2017468180\u2013186. (https:\/\/doi.org\/10.1016\/j.cca.2017.03.002)"},{"key":"bib15","series-title":"Clinica Chimica Acta","first-page":"6","article-title":"Sex, age, pubertal development and use of oral contraceptives in relation to serum concentrations of DHEA, DHEAS, 17\u03b1-hydroxyprogesterone, \u03944-androstenedione, testosterone and their ratios in children, adolescents and young adults","volume":"437","author":"S\u00f8eborg","year":"2014","unstructured":"S\u00f8eborg TFrederiksen HMouritsen AJohannsen THMain KMJ\u00f8rgensen NPetersen JHAndersson AM & Juul A. Sex, age, pubertal development and use of oral contraceptives in relation to serum concentrations of DHEA, DHEAS, 17\u03b1-hydroxyprogesterone, \u03944-androstenedione, testosterone and their ratios in children, adolescents and young adults. Clinica Chimica Acta20144376\u201313. (https:\/\/doi.org\/10.1016\/j.cca.2014.06.018)"},{"key":"bib16","series-title":"Clinica Chimica Acta","first-page":"82","article-title":"Reference ranges of 17-hydroxyprogesterone, DHEA, DHEAS, androstenedione, total and free testosterone determined by TurboFlow-LC-MS\/MS and associations to health markers in 304 men","volume":"454","author":"Damgaard-Olesen","year":"2016","unstructured":"Damgaard-Olesen AJohannsen THHolmboe SAS\u00f8eborg TPetersen JHAndersson AMAadahl MLinneberg A & Juul A. Reference ranges of 17-hydroxyprogesterone, DHEA, DHEAS, androstenedione, total and free testosterone determined by TurboFlow-LC-MS\/MS and associations to health markers in 304 men. Clinica Chimica Acta201645482\u201388. (https:\/\/doi.org\/10.1016\/j.cca.2015.12.042)"},{"key":"bib17","series-title":"European Journal of Endocrinology","first-page":"P1","article-title":"Peptide hormone analysis in diagnosis and treatment of Differences of Sex Development: joint position paper of EU COST Action \u2018DSDnet\u2019 and European Reference Network on Rare Endocrine Conditions","volume":"182","author":"Johannsen","year":"2020","unstructured":"Johannsen THAndersson AMAhmed SFRijke de YBGreaves RFHartmann MFHiort OHolterhus PMKrone NPKulle A, et al. Peptide hormone analysis in diagnosis and treatment of Differences of Sex Development: joint position paper of EU COST Action \u2018DSDnet\u2019 and European Reference Network on Rare Endocrine Conditions. European Journal of Endocrinology2020182P1\u2013P15. (https:\/\/doi.org\/10.1530\/EJE-19-0831)"},{"key":"bib18","series-title":"Acta Paediatrica","first-page":"214","article-title":"The 2014 Danish references from birth to 20 years for height, weight and body mass index","volume":"103","author":"Tinggaard","year":"2014","unstructured":"Tinggaard JAksglaede LS\u00f8rensen KMouritsen AWohlfahrt-Veje CHagen CPMieritz MGJ\u00f8rgensen NWolthers ODHeuck C, et al. The 2014 Danish references from birth to 20 years for height, weight and body mass index. Acta Paediatrica2014103214\u2013224. (https:\/\/doi.org\/10.1111\/apa.12468)"},{"key":"bib19","series-title":"Annals of Human Biology","first-page":"680","article-title":"References for growth and pubertal development from birth to 21 years in Flanders, Belgium","volume":"36","author":"Roelants","year":"2009","unstructured":"Roelants MHauspie R & Hoppenbrouwers K. References for growth and pubertal development from birth to 21 years in Flanders, Belgium. Annals of Human Biology200936680\u2013694. (https:\/\/doi.org\/10.3109\/03014460903049074)"},{"key":"bib20","series-title":"Journal of Clinical Endocrinology and Metabolism","first-page":"4043","article-title":"Congenital adrenal hyperplasia due to steroid 21-hydroxylase deficiency: an Endocrine Society clinical practice guideline","volume":"103","author":"Speiser","year":"2018","unstructured":"Speiser PWArlt WAuchus RJBaskin LSConway GSMerke DPMeyer-Bahlburg HFLMiller WLMurad MHOberfield SE, et al. Congenital adrenal hyperplasia due to steroid 21-hydroxylase deficiency: an Endocrine Society clinical practice guideline. Journal of Clinical Endocrinology and Metabolism20181034043\u20134088. (https:\/\/doi.org\/10.1210\/jc.2018-01865)"},{"key":"bib21","series-title":"European Journal of Endocrinology","first-page":"285","article-title":"Gonadal function in adult male patients with congenital adrenal hyperplasia","volume":"178","author":"Engels","year":"2018","unstructured":"Engels MGehrmann KFalhammar HWebb EANordenstr\u00f6m ASweep FCSpan PNHerwaarden van AERohayem JRichter-Unruh A, et al. Gonadal function in adult male patients with congenital adrenal hyperplasia. European Journal of Endocrinology2018178285\u2013294. (https:\/\/doi.org\/10.1530\/EJE-17-0862)"},{"key":"bib22","series-title":"Biology of Reproduction","first-page":"147","article-title":"INSL3 as a biomarker of Leydig cell functionality","volume":"88","author":"Ivell","year":"2013","unstructured":"Ivell RWade JD & Anand-Ivell R. INSL3 as a biomarker of Leydig cell functionality. Biology of Reproduction201388147. (https:\/\/doi.org\/10.1095\/biolreprod.113.108969)"},{"key":"bib23","series-title":"Journal of Clinical Endocrinology and Metabolism","article-title":"Serum insulin-like factor 3 levels are reduced in former androgen users, suggesting impaired Leydig cell capacity","volume":"106","author":"Rasmussen","year":"2021","unstructured":"Rasmussen JJAlbrethsen JFrandsen MNJ\u00f8rgensen NJuul A & Kistorp C. Serum insulin-like factor 3 levels are reduced in former androgen users, suggesting impaired Leydig cell capacity. Journal of Clinical Endocrinology and Metabolism2021106 e2664\u2013e2672. (https:\/\/doi.org\/10.1210\/clinem\/dgab129)"},{"key":"bib24","series-title":"Journal of Clinical Endocrinology and Metabolism","first-page":"3410","article-title":"Insulin-like factor 3 serum levels in 135 normal men and 85 men with testicular disorders: relationship to the luteinizing hormone-testosterone axis","volume":"90","author":"Bay","year":"2005","unstructured":"Bay KHartung SIvell RSchumacher MJ\u00fcrgensen DJorgensen NHolm MSkakkebaek NE & Andersson AM. Insulin-like factor 3 serum levels in 135 normal men and 85 men with testicular disorders: relationship to the luteinizing hormone-testosterone axis. Journal of Clinical Endocrinology and Metabolism2005903410\u20133418. (https:\/\/doi.org\/10.1210\/jc.2004-2257)"},{"key":"bib25","series-title":"Journal of Clinical Endocrinology and Metabolism","first-page":"2873","article-title":"Serum inhibin B and follicle-stimulating hormone levels as tools in the evaluation of infertile men: significance of adequate reference values from proven fertile men","volume":"89","author":"Andersson","year":"2004","unstructured":"Andersson AMPetersen JHJ\u00f8rgensen NJensen TK & Skakkeb\u00e6k NE. Serum inhibin B and follicle-stimulating hormone levels as tools in the evaluation of infertile men: significance of adequate reference values from proven fertile men. Journal of Clinical Endocrinology and Metabolism2004892873\u20132879. (https:\/\/doi.org\/10.1210\/jc.2003-032148)"},{"key":"bib26","series-title":"Fertility and Sterility","first-page":"2128","article-title":"Serum inhibin-b in fertile men is strongly correlated with low but not high sperm counts: a coordinated study of 1,797 European and US men","volume":"94","author":"J\u00f8rgensen","year":"2010","unstructured":"J\u00f8rgensen NLiu FAndersson AMVierula MIrvine DSAuger JBrazil CKDrobnis EZJensen TKJouannet P, et al. Serum inhibin-b in fertile men is strongly correlated with low but not high sperm counts: a coordinated study of 1,797 European and US men. Fertility and Sterility2010942128\u20132134. (https:\/\/doi.org\/10.1016\/j.fertnstert.2009.12.051)"},{"key":"bib27","series-title":"European Journal of Endocrinology","first-page":"331","article-title":"Bone mineral density, bone markers, and fractures in adult males with congenital adrenal hyperplasia","volume":"168","author":"Falhammar","year":"2013","unstructured":"Falhammar HFilipsson Nystr\u00f6m HWedell ABrismar K & Thor\u00e9n M. Bone mineral density, bone markers, and fractures in adult males with congenital adrenal hyperplasia. European Journal of Endocrinology2013168331\u2013341. (https:\/\/doi.org\/10.1530\/EJE-12-0865)"},{"key":"bib28","series-title":"Journal of Clinical Endocrinology and Metabolism","first-page":"E1547","article-title":"Relationship between final height and health outcomes in adults with congenital adrenal hyperplasia: United Kingdom Congenital Adrenal Hyperplasia Adult Study Executive (CaHASE","volume":"99","author":"Han","year":"2014","unstructured":"Han TSConway GSWillis DSKrone NRees DAStimson RHArlt WWalker BRRoss RJ & United Kingdom Congenital Adrenal Hyperplasia Adult Study Executive (CaHASE) Relationship between final height and health outcomes in adults with congenital adrenal hyperplasia: United Kingdom Congenital Adrenal Hyperplasia Adult Study Executive (CaHASE) Journal of Clinical Endocrinology and Metabolism201499E1547\u2013E1555. (https:\/\/doi.org\/10.1210\/jc.2014-1486)"},{"key":"bib29","series-title":"Journal of Clinical Endocrinology and Metabolism","first-page":"E524","article-title":"Molecular characterization of testicular adrenal rest tumors in congenital adrenal hyperplasia: lesions with both adrenocortical and Leydig cell features","volume":"100","author":"Smeets","year":"2015","unstructured":"Smeets EEJWSpan PNHerwaarden van AEWevers RAHermus ARMMSweep FCGJ & Claahsen-van der Grinten HL. Molecular characterization of testicular adrenal rest tumors in congenital adrenal hyperplasia: lesions with both adrenocortical and Leydig cell features. Journal of Clinical Endocrinology and Metabolism2015100E524\u2013E530. (https:\/\/doi.org\/10.1210\/jc.2014-2036)"},{"key":"bib30","series-title":"European Journal of Endocrinology","first-page":"491","article-title":"Abundance of DLK1, differential expression of CYP11B1, CYP21A2 and MC2R, and lack of INSL3 distinguish testicular adrenal rest tumours from Leydig cell tumours","volume":"172","author":"Lottrup","year":"2015","unstructured":"Lottrup GNielsen JESkakkeb\u00e6k NEJuul A & Rajpert-De Meyts E. Abundance of DLK1, differential expression of CYP11B1, CYP21A2 and MC2R, and lack of INSL3 distinguish testicular adrenal rest tumours from Leydig cell tumours. European Journal of Endocrinology2015172491\u2013499. (https:\/\/doi.org\/10.1530\/EJE-14-0810)"}],"container-title":["Endocrine Connections"],"original-title":[],"link":[{"URL":"https:\/\/ec.bioscientifica.com\/view\/journals\/ec\/12\/8\/EC-23-0073.xml","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/ec.bioscientifica.com\/view\/journals\/ec\/12\/8\/EC-23-0073.xml","content-type":"text\/html","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/ec.bioscientifica.com\/view\/journals\/ec\/12\/8\/EC-23-0073.xml","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,7,20]],"date-time":"2026-07-20T13:46:23Z","timestamp":1784555183000},"score":1,"resource":{"primary":{"URL":"http:\/\/journals.bioscientifica.com\/ec\/article\/1202"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,5,31]]},"references-count":30,"journal-issue":{"issue":"8"},"URL":"https:\/\/doi.org\/10.1530\/ec-23-0073","relation":{},"ISSN":["2049-3614"],"issn-type":[{"value":"2049-3614","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,5,31]]}}}