{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,30]],"date-time":"2025-10-30T17:16:21Z","timestamp":1761844581658,"version":"3.40.4"},"reference-count":10,"publisher":"BMJ","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Reports"],"accepted":{"date-parts":[[2016,2,19]]},"abstract":"<jats:p>Female hypospadias is a rare anomaly of the female urethra where it opens on the anterior vaginal wall anywhere between the introitus and the fornix. It is often associated with other genitourinary anomalies such as Cloacal malformation, female pseudohermaphroditism, nonneurogenic neurogenic bladder and urethral duplication. Idiopathic female pseudohermaphroditism is extremely rare, and most cases occur secondary to adrenogenital syndrome or maternal androgen exposure. We report a unique case of a 1-year and 4-month-old girl who presented with ambiguous genitalia and renal failure secondary to a non-neurogenic neurogenic bladder. On further evaluation, she was found to have urethral duplication with a hypospadiac female urethra. She initially underwent a vesicostomy and was further planned to undergo an appendicular Mitrofanoff at an older age. The mainstay of treatment in these cases includes relief of bladder outlet obstruction and recovery of renal function by adequate urinary drainage. Clitoral reduction, if cosmetically warranted, may be planned at puberty.<\/jats:p>","DOI":"10.1136\/bcr-2015-214172","type":"journal-article","created":{"date-parts":[[2016,3,10]],"date-time":"2016-03-10T16:09:09Z","timestamp":1457626149000},"page":"bcr-2015-214172","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":4,"title":["Idiopathic female pseudohermaphroditism with urethral duplication and female hypospadias"],"prefix":"10.1136","volume":"2016","author":[{"given":"Aureen Ruby","family":"D'Cunha","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jujju Jacob","family":"Kurian","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tarun John K","family":"Jacob","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2016,3,10]]},"reference":[{"key":"2025042702562475000_2016.mar10_1.bcr-2015-214172.1","doi-asserted-by":"crossref","first-page":"915","DOI":"10.1016\/S0022-5347(17)45127-5","article-title":"Vaginal and phallic urethra with prominent clitoris in female pseudo hermaphroditism","volume":"136","author":"Kounami","year":"1986","journal-title":"J Urol"},{"key":"2025042702562475000_2016.mar10_1.bcr-2015-214172.2","unstructured":"Jones HW , Scott WW Jr . Hermaphroditism, genital anomalies and related endocrine disorders. 2nd edn. Baltimore: The Williams and Wilkins Co, 1971:274."},{"key":"2025042702562475000_2016.mar10_1.bcr-2015-214172.3","first-page":"100","article-title":"Special female hermaphroditism associated with multiple disorders","volume":"39","author":"Park","year":"1972","journal-title":"Obstet Gynecol"},{"issue":"(Suppl)","key":"2025042702562475000_2016.mar10_1.bcr-2015-214172.4","first-page":"87","article-title":"Female pseudohermaphroditism","volume":"34","author":"Bierich","year":"1974","journal-title":"Helv Paediatr Acta"},{"key":"2025042702562475000_2016.mar10_1.bcr-2015-214172.5","doi-asserted-by":"publisher","DOI":"10.1097\/00003081-197709000-00006"},{"key":"2025042702562475000_2016.mar10_1.bcr-2015-214172.6","doi-asserted-by":"publisher","DOI":"10.1001\/archpedi.1958.02060050011002"},{"key":"2025042702562475000_2016.mar10_1.bcr-2015-214172.7","doi-asserted-by":"publisher","DOI":"10.1159\/000280952"},{"key":"2025042702562475000_2016.mar10_1.bcr-2015-214172.8","doi-asserted-by":"crossref","first-page":"805","DOI":"10.1016\/S0022-5347(17)56614-8","article-title":"Idiopathic female intersex with clitoromegaly and urethral duplication","volume":"122","author":"Belis","year":"1979","journal-title":"J Urol"},{"key":"2025042702562475000_2016.mar10_1.bcr-2015-214172.9","doi-asserted-by":"crossref","first-page":"1159","DOI":"10.1016\/S0022-5347(17)54280-9","article-title":"Accessory phallic urethra in the female patient","volume":"127","author":"Bellinger","year":"1982","journal-title":"J Urol"},{"key":"2025042702562475000_2016.mar10_1.bcr-2015-214172.10","first-page":"454","article-title":"Female pseudohermaphroditism: strategy and bias in a fast diagnosis. How tricky could be a diagnosis with a wrong anamnesis","volume":"85","author":"Onesti","year":"2014","journal-title":"Ann Ital Chir"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2015-214172","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,4,27]],"date-time":"2025-04-27T09:56:32Z","timestamp":1745747792000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2015-214172"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2016,3,10]]},"references-count":10,"alternative-id":["10.1136\/bcr-2015-214172"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2015-214172","relation":{},"ISSN":["1757-790X"],"issn-type":[{"type":"electronic","value":"1757-790X"}],"subject":[],"published":{"date-parts":[[2016,3,10]]}}}