{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,18]],"date-time":"2026-07-18T00:51:39Z","timestamp":1784335899245,"version":"3.55.0"},"reference-count":26,"publisher":"Elsevier BV","issue":"3","license":[{"start":{"date-parts":[[2017,6,1]],"date-time":"2017-06-01T00:00:00Z","timestamp":1496275200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2017,6,1]],"date-time":"2017-06-01T00:00:00Z","timestamp":1496275200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"}],"content-domain":{"domain":["clinicalkey.com","clinicalkey.com.au","clinicalkey.es","clinicalkey.fr","clinicalkey.jp","jpurol.com","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Journal of Pediatric Urology"],"published-print":{"date-parts":[[2017,6]]},"DOI":"10.1016\/j.jpurol.2017.01.004","type":"journal-article","created":{"date-parts":[[2017,1,30]],"date-time":"2017-01-30T14:17:22Z","timestamp":1485785842000},"page":"293.e1-293.e6","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":9,"title":["Variation in the clinical and genetic evaluation of undervirilized boys with bifid scrotum and hypospadias"],"prefix":"10.1016","volume":"13","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1017-9318","authenticated-orcid":false,"given":"J.M.","family":"Swartz","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"R.","family":"Ciarlo","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"E.","family":"Denhoff","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A.","family":"Abrha","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"D.A.","family":"Diamond","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J.N.","family":"Hirschhorn","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Y.-M.","family":"Chan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"key":"10.1016\/j.jpurol.2017.01.004_bib1","doi-asserted-by":"crossref","first-page":"672","DOI":"10.1016\/j.jpurol.2014.01.024","article-title":"Do adult men with untreated hypospadias have adverse outcomes? A pilot study using a social media advertised survey","volume":"10","author":"Schlomer","year":"2014","journal-title":"J Pediatr Urol"},{"key":"10.1016\/j.jpurol.2017.01.004_bib2","doi-asserted-by":"crossref","first-page":"463","DOI":"10.1016\/j.jpedsurg.2005.11.059","article-title":"Hypospadias: anatomy, etiology, and technique","volume":"41","author":"Baskin","year":"2006","journal-title":"J Pediatr Surg"},{"key":"10.1016\/j.jpurol.2017.01.004_bib3","doi-asserted-by":"crossref","first-page":"980121","DOI":"10.1155\/2015\/980121","article-title":"Parental reports of stigma associated with child's disorder of sex development","volume":"2015","author":"Rolston","year":"2015","journal-title":"Int J Endocrinol"},{"key":"10.1016\/j.jpurol.2017.01.004_bib4","doi-asserted-by":"crossref","first-page":"148","DOI":"10.1016\/j.jpurol.2006.03.004","article-title":"Consensus statement on management of intersex disorders","volume":"2","author":"Hughes","year":"2006","journal-title":"J Pediatr Urol"},{"key":"10.1016\/j.jpurol.2017.01.004_bib5","doi-asserted-by":"crossref","first-page":"569","DOI":"10.1016\/j.beem.2015.04.005","article-title":"Androgen insensitivity syndrome","volume":"29","author":"Mongan","year":"2015","journal-title":"Best Pract Res Clin Endocrinol Metab"},{"key":"10.1016\/j.jpurol.2017.01.004_bib6","doi-asserted-by":"crossref","first-page":"1419","DOI":"10.1016\/S0140-6736(12)60071-3","article-title":"Androgen insensitivity syndrome","volume":"380","author":"Hughes","year":"2012","journal-title":"Lancet"},{"key":"10.1016\/j.jpurol.2017.01.004_bib7","first-page":"158","article-title":"Genetic testing practices in infants with congenital","author":"Connor","year":"2013","journal-title":"Congenit Hear Dis"},{"key":"10.1016\/j.jpurol.2017.01.004_bib8","doi-asserted-by":"crossref","first-page":"1674","DOI":"10.1016\/S0140-6736(12)61480-9","article-title":"Range of genetic mutations associated with severe non-syndromic sporadic intellectual disability: an exome sequencing study","volume":"380","author":"Rauch","year":"2012","journal-title":"Lancet"},{"key":"10.1016\/j.jpurol.2017.01.004_bib9","doi-asserted-by":"crossref","first-page":"451","DOI":"10.1016\/j.ymgme.2014.12.304","article-title":"Molecular genetic testing of patients with monogenic diabetes and hyperinsulinism","volume":"114","author":"Bennett","year":"2015","journal-title":"Mol Genet Metab"},{"key":"10.1016\/j.jpurol.2017.01.004_bib10","doi-asserted-by":"crossref","first-page":"E333","DOI":"10.1210\/jc.2014-2605","article-title":"Exome sequencing for the diagnosis of 46,XY disorders of sex development","volume":"100","author":"Baxter","year":"2015","journal-title":"J Clin Endocrinol Metab"},{"key":"10.1016\/j.jpurol.2017.01.004_bib11","doi-asserted-by":"crossref","first-page":"35","DOI":"10.1111\/j.1399-0004.2012.01879.x","article-title":"Targeted massively parallel sequencing provides comprehensive genetic diagnosis for patients with disorders of sex development","volume":"83","author":"Arboleda","year":"2013","journal-title":"Clin Genet"},{"key":"10.1016\/j.jpurol.2017.01.004_bib12","first-page":"312","article-title":"New genomic technologies: an aid for diagnosis of disorders of sex development","author":"Barseghyan","year":"2015","journal-title":"Horm Metab Res"},{"key":"10.1016\/j.jpurol.2017.01.004_bib13","doi-asserted-by":"crossref","first-page":"603","DOI":"10.1038\/nrendo.2014.130","article-title":"DSDs: genetics, underlying pathologies and psychosexual differentiation","volume":"10","author":"Arboleda","year":"2014","journal-title":"Nat Rev Endocrinol"},{"key":"10.1016\/j.jpurol.2017.01.004_bib14","doi-asserted-by":"crossref","first-page":"209","DOI":"10.1186\/s13023-014-0209-2","article-title":"Extensive clinical, hormonal and genetic screening in a large consecutive series of 46,XY neonates and infants with atypical sexual development","volume":"9","author":"Baetens","year":"2014","journal-title":"Orphanet J Rare Dis"},{"key":"10.1016\/j.jpurol.2017.01.004_bib15","doi-asserted-by":"crossref","first-page":"206","DOI":"10.1016\/j.adnc.2004.05.003","article-title":"Hypospadias in the neonate","volume":"4","author":"Stokowski","year":"2004","journal-title":"Adv Neonatal Care"},{"key":"10.1016\/j.jpurol.2017.01.004_bib16","doi-asserted-by":"crossref","first-page":"79","DOI":"10.1038\/nrendo.2012.235","article-title":"Disorders of sex development: new genes, new concepts","volume":"9","author":"Ono","year":"2013","journal-title":"Nat Rev Endocrinol"},{"key":"10.1016\/j.jpurol.2017.01.004_bib17","first-page":"1040","article-title":"Integration of clinical and genetic data in the i2b2 architecture","author":"Murphy","year":"2006","journal-title":"AMIA Annu Symp Proc"},{"key":"10.1016\/j.jpurol.2017.01.004_bib18","doi-asserted-by":"crossref","first-page":"377","DOI":"10.1016\/j.jbi.2008.08.010","article-title":"Research Electronic Data Capture (REDCap) \u2013 a metadata driven methodology and workflow process for providing translational research informatics support","volume":"42","author":"Harris","year":"2009","journal-title":"J Biomed Inf"},{"key":"10.1016\/j.jpurol.2017.01.004_bib19","doi-asserted-by":"crossref","first-page":"2183","DOI":"10.1002\/ajmg.a.36050","article-title":"Severe hypospadias and its association with maternal-placental factors","volume":"161","author":"Huisma","year":"2013","journal-title":"Am J Med Genet"},{"key":"10.1016\/j.jpurol.2017.01.004_bib20","doi-asserted-by":"crossref","first-page":"10","DOI":"10.1210\/jc.2006-1374","article-title":"Care of girls and women with Turner syndrome: a guideline of the Turner Syndrome Study Group","volume":"92","author":"Bondy","year":"2007","journal-title":"J Clin Endocrinol Metab"},{"key":"10.1016\/j.jpurol.2017.01.004_bib21","doi-asserted-by":"crossref","first-page":"1502","DOI":"10.1056\/NEJMoa1306555","article-title":"Clinical whole-exome sequencing for the diagnosis of Mendelian disorders","volume":"369","author":"Yang","year":"2013","journal-title":"N Engl J Med"},{"key":"10.1016\/j.jpurol.2017.01.004_bib22","doi-asserted-by":"crossref","first-page":"607","DOI":"10.1016\/j.beem.2015.07.004","article-title":"DAX-1 (NR0B1) and steroidogenic factor-1 (SF-1, NR5A1) in human disease","volume":"29","author":"Suntharalingham","year":"2015","journal-title":"Best Pract Res Clin Endocrinol Metab"},{"key":"10.1016\/j.jpurol.2017.01.004_bib23","article-title":"Two unrelated undervirilized 46,XY males with inherited NR5A1 variants identified by whole-exome sequencing","author":"Swartz","year":"2016","journal-title":"Horm Res Paediatr"},{"key":"10.1016\/j.jpurol.2017.01.004_bib24","doi-asserted-by":"crossref","first-page":"957","DOI":"10.1038\/ng.3063","article-title":"Genome-wide association analyses identify variants in developmental genes associated with hypospadias","volume":"46","author":"Geller","year":"2014","journal-title":"Nat Genet"},{"key":"10.1016\/j.jpurol.2017.01.004_bib25","doi-asserted-by":"crossref","first-page":"333","DOI":"10.1136\/jmg.23.4.333","article-title":"Aetiological factors in hypospadias","volume":"23","author":"Calzolari","year":"1986","journal-title":"J Med Genet"},{"key":"10.1016\/j.jpurol.2017.01.004_bib26","doi-asserted-by":"crossref","first-page":"1071","DOI":"10.1289\/ehp.10791","article-title":"Maternal and gestational risk factors for hypospadias","volume":"116","author":"Akre","year":"2008","journal-title":"Environ Health Perspect"}],"container-title":["Journal of Pediatric Urology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S1477513117300451?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S1477513117300451?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,10,26]],"date-time":"2025-10-26T13:42:57Z","timestamp":1761486177000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S1477513117300451"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2017,6]]},"references-count":26,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2017,6]]}},"alternative-id":["S1477513117300451"],"URL":"https:\/\/doi.org\/10.1016\/j.jpurol.2017.01.004","relation":{},"ISSN":["1477-5131"],"issn-type":[{"value":"1477-5131","type":"print"}],"subject":[],"published":{"date-parts":[[2017,6]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Variation in the clinical and genetic evaluation of undervirilized boys with bifid scrotum and hypospadias","name":"articletitle","label":"Article Title"},{"value":"Journal of Pediatric Urology","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.jpurol.2017.01.004","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2017 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved.","name":"copyright","label":"Copyright"}]}}