Laura Gallardo Zamora , Yesica Quiroz Madarriaga , Anna Bujons Tur , Antoni Sanchez i Puy , David Salinas Duffo , Juan Antonio Peña Gonzalez
{"title":"46，XX男性先天性肾上腺增生症（CAH） 21-羟化酶缺乏导致尿道狭窄：文献复习并病例报告","authors":"Laura Gallardo Zamora , Yesica Quiroz Madarriaga , Anna Bujons Tur , Antoni Sanchez i Puy , David Salinas Duffo , Juan Antonio Peña Gonzalez","doi":"10.1016/j.eucr.2025.103182","DOIUrl":null,"url":null,"abstract":"<div><div>Disorders of sex development in 46,XX male individuals are rare and present complex surgical challenges, particularly regarding urethral stricture following urethral surgery. We present a 46,XX individual raised male who underwent early urethral surgery and developed a urethral stricture in adulthood. Surgical repair was not feasible due to anatomy and comorbidities. A systematic review on the management of urethral strictures in this patient population was conducted. Only one case of successful buccal mucosa graft urethroplasty in a 46,XX male was found. This case underscores the scarcity of data and challenges in managing urethral strictures in virilized 46,XX individuals.</div></div>","PeriodicalId":38188,"journal":{"name":"Urology Case Reports","volume":"63 ","pages":"Article 103182"},"PeriodicalIF":0.4000,"publicationDate":"2025-08-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Urethral stricture in a 46,XX male with congenital adrenal hyperplasia (CAH) due to 21-hydroxylase deficiency: A literature review and case report\",\"authors\":\"Laura Gallardo Zamora , Yesica Quiroz Madarriaga , Anna Bujons Tur , Antoni Sanchez i Puy , David Salinas Duffo , Juan Antonio Peña Gonzalez\",\"doi\":\"10.1016/j.eucr.2025.103182\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<div><div>Disorders of sex development in 46,XX male individuals are rare and present complex surgical challenges, particularly regarding urethral stricture following urethral surgery. We present a 46,XX individual raised male who underwent early urethral surgery and developed a urethral stricture in adulthood. Surgical repair was not feasible due to anatomy and comorbidities. A systematic review on the management of urethral strictures in this patient population was conducted. Only one case of successful buccal mucosa graft urethroplasty in a 46,XX male was found. This case underscores the scarcity of data and challenges in managing urethral strictures in virilized 46,XX individuals.</div></div>\",\"PeriodicalId\":38188,\"journal\":{\"name\":\"Urology Case Reports\",\"volume\":\"63 \",\"pages\":\"Article 103182\"},\"PeriodicalIF\":0.4000,\"publicationDate\":\"2025-08-27\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Urology Case Reports\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://www.sciencedirect.com/science/article/pii/S2214442025002530\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q4\",\"JCRName\":\"UROLOGY & NEPHROLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Urology Case Reports","FirstCategoryId":"1085","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S2214442025002530","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q4","JCRName":"UROLOGY & NEPHROLOGY","Score":null,"Total":0}
Urethral stricture in a 46,XX male with congenital adrenal hyperplasia (CAH) due to 21-hydroxylase deficiency: A literature review and case report
Disorders of sex development in 46,XX male individuals are rare and present complex surgical challenges, particularly regarding urethral stricture following urethral surgery. We present a 46,XX individual raised male who underwent early urethral surgery and developed a urethral stricture in adulthood. Surgical repair was not feasible due to anatomy and comorbidities. A systematic review on the management of urethral strictures in this patient population was conducted. Only one case of successful buccal mucosa graft urethroplasty in a 46,XX male was found. This case underscores the scarcity of data and challenges in managing urethral strictures in virilized 46,XX individuals.
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