{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,29]],"date-time":"2026-10-29T20:14:15Z","timestamp":1793304855294,"version":"4.5.0"},"reference-count":77,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2022,7,14]],"date-time":"2022-07-14T00:00:00Z","timestamp":1657756800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100014013","name":"UK Research and Innovation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100014013","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000265","name":"Medical Research Council","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000265","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000589","name":"Chief Scientist Office","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000589","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Endocrinol."],"abstract":"The Sertoli cells of the testes play an essential role during gonadal development, in addition to supporting subsequent germ cell survival and spermatogenesis. Anti-M\u00fcllerian hormone (AMH) is a member of the TGF-\u03b2 superfamily, which is secreted by immature Sertoli cells from the 8th<\/jats:sup> week of fetal gestation. lnhibin B is a glycoprotein, which is produced by the Sertoli cells from early in fetal development. In people with a Difference or Disorder of Sex Development (DSD), these hormones may be useful to determine the presence of testicular tissue and potential for spermatogenesis. However, fetal Sertoli cell development and function is often dysregulated in DSD conditions and altered production of Sertoli cell hormones may be detected throughout the life course in these individuals. As such this review will consider the role of AMH and inhibin B in individuals with DSD.<\/jats:p>","DOI":"10.3389\/fendo.2022.919670","type":"journal-article","created":{"date-parts":[[2022,7,14]],"date-time":"2022-07-14T05:53:45Z","timestamp":1657778025000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":25,"title":["Testicular Sertoli Cell Hormones in Differences in Sex Development"],"prefix":"10.3389","volume":"13","author":[{"given":"Angela K.","family":"Lucas-Herald","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rod T.","family":"Mitchell","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2022,7,14]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","first-page":"189","DOI":"10.1111\/andr.12165","article-title":"The Sertoli Cell: One Hundred Fifty Years of Beauty and Plasticity","volume":"4","author":"Fran\u00e7a","year":"2016","journal-title":"Andrology"},{"key":"B2","first-page":"905232","article-title":"Sexual Differentiation","volume-title":"Endotext","author":"Rey","year":"2016"},{"key":"B3","doi-asserted-by":"publisher","first-page":"764","DOI":"10.1016\/j.stem.2020.12.004","article-title":"Single-Cell Analysis of the Developing Human Testis Reveals Somatic Niche Cell Specification and Fetal Germline Stem Cell Establishment","volume":"28","author":"Guo","year":"2021","journal-title":"Cell Stem Cell"},{"key":"B4","article-title":"Chapter Six - Characterizing the Bipotential Mammalian Gonad","volume-title":"Curr Top Dev Biol","author":"Nef","year":"2019"},{"key":"B5","doi-asserted-by":"publisher","DOI":"10.1159\/000519217","article-title":"Transcriptional Regulation of the Y-Linked Mammalian Testis-Determining Gene SRY","volume":"15","author":"Okashita","year":"2021","journal-title":"Sexual Dev"},{"key":"B6","doi-asserted-by":"publisher","DOI":"10.1007\/s10577-011-9256-x","article-title":"SRY Protein Function in Sex Determination: Thinking Outside the Box","volume":"20","author":"Zhao","year":"2012","journal-title":"Chromosome Res"},{"key":"B7","doi-asserted-by":"publisher","DOI":"10.1038\/nature06944","article-title":"Sex Determination Involves Synergistic Action of SRY and SF1 on a Specific Sox9 Enhancer","volume":"453","author":"Sekido","year":"2008","journal-title":"Nature"},{"key":"B8","doi-asserted-by":"publisher","first-page":"15961","DOI":"10.1038\/s41598-017-15931-3","article-title":"Temporal Expression Pattern of Genes During the Period of Sex Differentiation in Human Embryonic Gonads","volume":"7","author":"Mamsen","year":"2017","journal-title":"Sci Rep"},{"key":"B9","doi-asserted-by":"publisher","DOI":"10.1016\/S0925-4773(99)00123-9","article-title":"Expression of Steroidogenic Factor 1 and Wilms' Tumour 1 During Early Human Gonadal Development and Sex Determination","volume":"87","author":"Hanley","year":"1999","journal-title":"Mech Dev"},{"key":"B10","doi-asserted-by":"publisher","DOI":"10.1101\/gad.981202","article-title":"Desert Hedgehog\/Patched 1 Signaling Specifies Fetal Leydig Cell Fate in Testis Organogenesis","volume":"16","author":"Yao","year":"2002","journal-title":"Genes Dev"},{"key":"B11","doi-asserted-by":"publisher","first-page":"96","DOI":"10.1016\/j.ydbio.2009.02.025","article-title":"Activation of the Hedgehog Pathway in the Mouse Fetal Ovary Leads to Ectopic Appearance of Fetal Leydig Cells and Female Pseudohermaphroditism","volume":"329","author":"Barsoum","year":"2009","journal-title":"Dev Biol"},{"key":"B12","volume-title":"The Developing Human: Clinically Orientated Embryology","author":"Moore","year":"2016"},{"key":"B13","doi-asserted-by":"publisher","first-page":"17","DOI":"10.1159\/000363609","article-title":"The Masculinization Programming Window","volume":"27","author":"Welsh","year":"2014","journal-title":"Endocr Dev"},{"key":"B14","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpedsurg.2013.02.001","article-title":"Undescended Testis: The Underlying Mechanisms and the Effects on Germ Cells That Cause Infertility and Cancer","volume":"48","author":"Hutson","year":"2013","journal-title":"J Pediatr Surg"},{"key":"B15","doi-asserted-by":"publisher","first-page":"20","DOI":"10.1186\/s13633-016-0038-2","article-title":"Anti-M\u00fcllerian Hormone as a Marker of Steroid and Gonadotropin Action in the Testis of Children and Adolescents With Disorders of the Gonadal Axis","volume":"2016","author":"Edelsztein","year":"2016","journal-title":"Int J Pediatr Endocrinol"},{"key":"B16","doi-asserted-by":"publisher","first-page":"2","DOI":"10.1159\/000132332","article-title":"Mapping of the Gene for Anti-M\u00fcllerian Hormone to the Short Arm of Human Chromosome 19","volume":"44","author":"Cohen-Haguenauer","year":"1987","journal-title":"Cytogenet Genome Res"},{"key":"B17","doi-asserted-by":"publisher","DOI":"10.1152\/ajpendo.00187.2011","article-title":"SOX9 and SF1 are Involved in Cyclic AMP-Mediated Upregulation of Anti-M\u00fcllerian Gene Expression in the Testicular Prepubertal Sertoli Cell Line SMAT1","volume":"301","author":"Lasala","year":"2011","journal-title":"Am J Physiol Endocrinol Metab"},{"key":"B18","doi-asserted-by":"publisher","DOI":"10.3389\/fendo.2020.00619","article-title":"What Does AMH Tell Us in Pediatric Disorders of Sex Development","volume":"11","author":"Josso","year":"2020","journal-title":"Front Endocrinol"},{"key":"B19","doi-asserted-by":"publisher","first-page":"101634","DOI":"10.1016\/j.beem.2022.101634","article-title":"Genetics of Anti-M\u00fcllerian Hormone and Its Signaling Pathway","volume":"36","author":"Josso","year":"2022","journal-title":"Best Pract Res Clin Endocrinol Metab"},{"key":"B20","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2008-0915","article-title":"Physiological Androgen Insensitivity of the Fetal, Neonatal, and Early Infantile Testis Is Explained by the Ontogeny of the Androgen Receptor Expression in Sertoli Cells","volume":"93","author":"Chemes","year":"2008","journal-title":"J Clin Endocrinol Metab"},{"key":"B21","doi-asserted-by":"publisher","DOI":"10.1093\/biolre\/ioy152","article-title":"Androgens Downregulate Anti-M\u00fcllerian Hormone Promoter Activity in the Sertoli Cell Through the Androgen Receptor and Intact Steroidogenic Factor 1 Sites","volume":"99","author":"Edelsztein","year":"2018","journal-title":"Biol Reprod"},{"key":"B22","doi-asserted-by":"publisher","DOI":"10.1093\/humupd\/dmh057","article-title":"Inhibins in Female and Male Reproductive Physiology: Role in Gametogenesis, Conception, Implantation and Early Pregnancy","volume":"11","author":"Luisi","year":"2005","journal-title":"Hum Reprod Update"},{"key":"B23","doi-asserted-by":"publisher","DOI":"10.1038\/s10038-020-0757-3","article-title":"Genome-Wide Association Study of Semen Volume, Sperm Concentration, Testis Size, and Plasma Inhibin B Levels","volume":"65","author":"Sato","year":"2020","journal-title":"J Hum Genet"},{"key":"B24","doi-asserted-by":"publisher","first-page":"94","DOI":"10.1002\/j.1939-4640.1999.tb02501.x","article-title":"Inhibin B Levels in Plasma of the Male Rat From Birth to Adulthood: Effect of Experimental Manipulation of Sertoli Cell Number","volume":"20","author":"Sharpe","year":"1999","journal-title":"J Androl"},{"key":"B25","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2605.2000.00229.x","article-title":"Regulation of Inhibin Production in the Human Male and Its Clinical Applications","volume":"23","author":"Anderson","year":"2000","journal-title":"Int J Androl"},{"key":"B26","doi-asserted-by":"publisher","first-page":"883","DOI":"10.1210\/er.2009-0016","article-title":"Steroidogenesis in the Fetal Testis and Its Susceptibility to Disruption by Exogenous Compounds","volume":"30","author":"Scott","year":"2009","journal-title":"Endocr Rev"},{"key":"B27","doi-asserted-by":"publisher","DOI":"10.1016\/0378-3782(93)90204-8","article-title":"Anti-M\u00fcllerian Hormone in Early Human Development","volume":"33","author":"Josso","year":"1993","journal-title":"Early Hum Dev"},{"key":"B28","doi-asserted-by":"publisher","first-page":"33","DOI":"10.1016\/j.rbmo.2013.03.009","article-title":"Reproductive Hormone Concentrations in Pregnancy and Neonates: A Systematic Review","volume":"27","author":"Kuijper","year":"2013","journal-title":"Reprod BioMed Online"},{"key":"B29","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1159\/000518273","article-title":"AMH and AMHR2 Involvement in Congenital Disorders of Sex Development","volume":"31","author":"Brunello","year":"2021","journal-title":"Sexual Dev"},{"key":"B30","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2010-1207","article-title":"Changes in Anti-M\u00fcllerian Hormone (AMH) Throughout the Life Span: A Population-Based Study of 1027 Healthy Males From Birth (Cord Blood) to the Age of 69 Years","volume":"95","author":"Aksglaede","year":"2010","journal-title":"J Clin Endocrinol Metab"},{"key":"B31","doi-asserted-by":"publisher","DOI":"10.1007\/978-3-319-31973-5_9","article-title":"Biology of the Sertoli Cell in the Fetal, Pubertal, and Adult Mammalian Testis","volume":"58","author":"Chojnacka","year":"2016","journal-title":"Mol Mech Cell Differ Gonad Dev"},{"key":"B32","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2006-1079","article-title":"Time Course of the Serum Gonadotropin Surge, Inhibins, and Anti-Mullerian Hormone in Normal Newborn Males During the First Month of Life","volume":"91","author":"Bergad\u00e1","year":"2006","journal-title":"J Clin Endocrinol Metab"},{"key":"B33","doi-asserted-by":"publisher","DOI":"10.1016\/S1472-6483(10)61653-7","article-title":"In-Vivo Concentrations of Inhibins, Activin A and Follistatin in Human Early Pregnancy","volume":"8","author":"Muttukrishna","year":"2004","journal-title":"Reprod Biomed Online"},{"key":"B34","doi-asserted-by":"publisher","DOI":"10.1007\/s004310051309","article-title":"Serum Inhibin B in Normal Term-Born Male and Female Neonates During the First Week of Life","volume":"159","author":"De Schepper","year":"2000","journal-title":"Eur J Pediatr"},{"key":"B35","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2605.1987.tb00358.x","article-title":"Proliferation of Sertoli Cells During Development of the Human Testis Assessed by Stereological Methods","volume":"10","author":"Cortes","year":"1987","journal-title":"Int J Androl"},{"key":"B36","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0153843","article-title":"A Normative Model of Serum Inhibin B in Young Males","volume":"11","author":"Kelsey","year":"2016","journal-title":"PLoS One"},{"key":"B37","doi-asserted-by":"crossref","DOI":"10.1530\/EJE-19-0831","article-title":"Peptide hormone analysis in diagnosis and treatment of Differences of Sex Development: joint position paper of EU COST Action 'DSDnet' and European Reference Network on Rare Endocrine Conditions","volume":"182","author":"Johannsen","year":"2020","journal-title":"Eur J Endocrinol"},{"key":"B38","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2002-021491","article-title":"Prenatal and Postnatal Prevalence of Klinefelter Syndrome: A National Registry Study","volume":"88","author":"Bojesen","year":"2003","journal-title":"J Clin Endocrinol Metab"},{"key":"B39","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2005-0697","article-title":"Morbidity in Klinefelter Syndrome: A Danish Register Study Based on Hospital Discharge Diagnoses","volume":"91","author":"Bojesen","year":"2006","journal-title":"J Clin Endocrinol Metab"},{"key":"B40","doi-asserted-by":"publisher","DOI":"10.1111\/j.1651-2227.2011.02148.x","article-title":"Serum Concentrations of Anti-M\u00fcllerian Hormone (AMH) in 95 Patients With Klinefelter Syndrome With or Without Cryptorchidism","volume":"100","author":"Aksglaede","year":"2011","journal-title":"Acta Paediatr"},{"key":"B41","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2010-2103","article-title":"Assessment of Leydig and Sertoli Cell Functions in Infants With Nonmosaic Klinefelter Syndrome: Insulin-Like Peptide 3 Levels Are Normal and Positively Correlated With LH Levels","volume":"96","author":"Cabrol","year":"2011","journal-title":"J Clin Endocrinol Metab"},{"key":"B42","doi-asserted-by":"publisher","first-page":"67","DOI":"10.1093\/bmb\/ldt008","article-title":"Understanding the Genetic Aetiology in Patients With XY DSD","volume":"106","author":"Ahmed","year":"2013","journal-title":"Br Med Bull"},{"key":"B43","doi-asserted-by":"publisher","DOI":"10.1152\/physrev.00009.2006","article-title":"Sex Determination and Gonadal Development in Mammals","volume":"87","author":"Wilhelm","year":"2007","journal-title":"Physiol Rev"},{"key":"B44","doi-asserted-by":"publisher","first-page":"16","DOI":"10.2164\/jandrol.109.008201","article-title":"Determination and Stability of Gonadal Sex","volume":"31","author":"Schlessinger","year":"2010","journal-title":"J Androl"},{"key":"B45","doi-asserted-by":"publisher","DOI":"10.6065\/apem.2014.19.2.108","article-title":"A Korean Boy With 46,XX Testicular Disorder of Sex Development Caused by SOX9 Duplication","volume":"19","author":"Lee","year":"2014","journal-title":"Ann Pediatr Endocrinol Metab"},{"key":"B46","doi-asserted-by":"publisher","first-page":"825","DOI":"10.1136\/jmedgenet-2011-100255","article-title":"Disruption of a Long Distance Regulatory Region Upstream of ≪Em