{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,9,25]],"date-time":"2025-09-25T15:51:01Z","timestamp":1758815461307,"version":"3.40.5"},"reference-count":30,"publisher":"Oxford University Press (OUP)","issue":"5","license":[{"start":{"date-parts":[[2022,5,1]],"date-time":"2022-05-01T00:00:00Z","timestamp":1651363200000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["eje.bioscientifica.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2022,5,1]]},"abstract":"<jats:sec>\n<jats:title>Background<\/jats:title>\n<jats:p>The human <jats:italic>INHA<\/jats:italic> gene encodes the inhibin subunit alpha protein, which is common to both inhibin A and B. The functional importance of inhibins in male sex development, sexual function, and reproduction remain largely unknown.<\/jats:p>\n<\/jats:sec>\n<jats:sec>\n<jats:title>Objective<\/jats:title>\n<jats:p>We report for the first time two male siblings with homozygous <jats:italic>INHA<\/jats:italic>mutations.<\/jats:p>\n<\/jats:sec>\n<jats:sec>\n<jats:title>Methods<\/jats:title>\n<jats:p>The medical files were examined for clinical, biochemical, and imaging data. Genetic analysis was performed using next-generation and Sanger sequencing methods.<\/jats:p>\n<\/jats:sec>\n<jats:sec>\n<jats:title>Results<\/jats:title>\n<jats:p>Two brothers complained of gynecomastia, testicular pain, and had a history of hypospadias. Biochemistry revealed low serum testosterone, high gonadotropin and anti-Mullerian hormone, and very low\/undetectable inhibin concentrations, where available. Both patients had azoospermia in the spermiogram. We have identified a homozygous 2 bp deletion (c.208_209delAG, R70Gfs*3) variant, which leads to a truncated INHA protein in both patients, and confirmed heterozygosity in the parents. The external genital development, pubertal onset and progression, reproductive functions, serum gonadotropins, and sex hormones of mother and father, who were heterozygous carriers of the identified mutation, were normal.<\/jats:p>\n<\/jats:sec>\n<jats:sec>\n<jats:title>Conclusion<\/jats:title>\n<jats:p>Homozygosity for <jats:italic>INHA<\/jats:italic> mutations causes decreased prenatal and postnatal testosterone production and infertility in males, while the heterozygous female and male carriers of <jats:italic>INHA<\/jats:italic> mutations do not have any abnormality in sex development and reproduction.<\/jats:p>\n<\/jats:sec>","DOI":"10.1530\/eje-21-1230","type":"journal-article","created":{"date-parts":[[2022,3,2]],"date-time":"2022-03-02T19:48:20Z","timestamp":1646250500000},"page":"K25-K31","update-policy":"https:\/\/doi.org\/10.1530\/crossmarkpolicy-7","source":"Crossref","is-referenced-by-count":3,"title":["Homozygosity for a novel INHA mutation in two male siblings with hypospadias, primary hypogonadism, and high-normal testicular volume"],"prefix":"10.1093","volume":"186","author":[{"given":"Esra","family":"Arslan Ates","sequence":"first","affiliation":[{"name":"Marmara University Pendik Training and Research Hospital, Genetic Diseases Diagnostic Center, Istanbul, Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mehmet","family":"Eltan","sequence":"additional","affiliation":[{"name":"Department of Pediatric Endocrinology and Diabetes, Marmara University, School of Medicine, Istanbul, Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Bahadir","family":"Sahin","sequence":"additional","affiliation":[{"name":"Department of Urology, Marmara University, School of Medicine, Istanbul, Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Busra","family":"Gurpinar Tosun","sequence":"additional","affiliation":[{"name":"Department of Pediatric Endocrinology and Diabetes, Marmara University, School of Medicine, Istanbul, Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tuba","family":"Seven Menevse","sequence":"additional","affiliation":[{"name":"Department of Pediatric Endocrinology and Diabetes, Marmara University, School of Medicine, Istanbul, Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Bilgen Bilge","family":"Geckinli","sequence":"additional","affiliation":[{"name":"Marmara University Pendik Training and Research Hospital, Genetic Diseases Diagnostic Center, Istanbul, Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Andy","family":"Greenfield","sequence":"additional","affiliation":[{"name":"Mammalian Genetics Unit, MRC Harwell Institute, Oxfordshire, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Serap","family":"Turan","sequence":"additional","affiliation":[{"name":"Department of Pediatric Endocrinology and Diabetes, Marmara University, School of Medicine, Istanbul, Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Abdullah","family":"Bereket","sequence":"additional","affiliation":[{"name":"Department of Pediatric Endocrinology and Diabetes, Marmara University, School of Medicine, Istanbul, Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2658-6866","authenticated-orcid":true,"given":"Tulay","family":"Guran","sequence":"additional","affiliation":[{"name":"Department of Pediatric Endocrinology and Diabetes, Marmara University, School of Medicine, Istanbul, Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","reference":[{"key":"bib1","series-title":"Molecular Endocrinology","doi-asserted-by":"publisher","first-page":"561","DOI":"10.1210\/mend-1-8-561","article-title":"Rat inhibin: molecular cloning of alpha- and beta-subunit complementary deoxyribonucleic acids and expression in the ovary","volume":"1","author":"Woodruff","year":"1987","unstructured":"Woodruff TKMeunier HJones PBHsueh AJMayo KE. 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Mutational analysis of the mature peptide region of inhibin genes in Indian women with ovarian failure. Human Reproduction2004191760\u20131764. (https:\/\/doi.org\/10.1093\/humrep\/deh342)10.1093\/humrep\/deh342"},{"key":"bib14","series-title":"Genome Medicine","article-title":"The Human Gene Mutation Database: 2008 update","volume":"1","author":"Stenson","year":"2009","unstructured":"Stenson PDMort MBall EVHowells KPhillips ADThomas NSCooper DN. The Human Gene Mutation Database: 2008 update. Genome Medicine20091 13. (https:\/\/doi.org\/10.1186\/gm13)"},{"key":"bib15","series-title":"Nucleic Acids Research","article-title":"ClinVar: improving access to variant interpretations and supporting evidence","volume":"46","author":"Landrum","year":"2018","unstructured":"Landrum MJLee JMBenson MBrown GRChao CChitipiralla SGu BHart JHoffman DJang W. ClinVar: improving access to variant interpretations and supporting evidence. Nucleic Acids Research201846 D1062\u2013D1067. 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(https:\/\/doi.org\/10.1038\/ng.2892)10.1038\/ng.2892"},{"key":"bib21","series-title":"Genetics in Medicine","doi-asserted-by":"publisher","first-page":"405","DOI":"10.1038\/gim.2015.30","article-title":"Standards and guidelines for the interpretation of sequence variants: a joint consensus recommendation of the American College of Medical Genetics and Genomics and the Association for Molecular Pathology","volume":"17","author":"Richards","year":"2015","unstructured":"Richards SAziz NBale SBick DDas SGastier-Foster JGrody WWHegde MLyon ESpector EStandards and guidelines for the interpretation of sequence variants: a joint consensus recommendation of the American College of Medical Genetics and Genomics and the Association for Molecular Pathology. Genetics in Medicine201517405\u2013424. 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Effect of inhibin on rat testicular desoxyribonucleic acid (DNA) synthesis in vivo and in vitro. Acta Endocrinologica198198312\u2013320. (https:\/\/doi.org\/10.1530\/acta.0.0980312)10.1530\/acta.0.0980312"},{"key":"bib25","series-title":"Endocrinology","first-page":"1899","article-title":"Inhibin reduces spermatogonial numbers in testes of adult mice and Chinese hamsters","volume":"125","author":"van Dissel-Emiliani","year":"1989","unstructured":"van Dissel-Emiliani FMGrootenhuis AJde Jong FHde Rooij DG. Inhibin reduces spermatogonial numbers in testes of adult mice and Chinese hamsters. Endocrinology19891251899\u20131903. 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