{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,24]],"date-time":"2026-08-24T06:19:17Z","timestamp":1787552357664,"version":"build-2736575974"},"reference-count":26,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2026,8,24]],"date-time":"2026-08-24T00:00:00Z","timestamp":1787529600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Pediatr."],"abstract":"<jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>To present a case series of the proximal hypospadias associated with perineal mass, which the perineal lesions were lined by urothelium containing Von Brunn's nests. A literature review was conducted to summarize the clinical and pathological features of similar cases.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Two cases of proximal hypospadias with perineal mass treated at our institute were analyzed. Clinical presentation, diagnostic evaluation, surgical management, and histopathological findings are described. A literature review was performed in PubMed with the terms \u201c(\u2018perineal mass\u2019 OR \u2018perineal anomaly\u2019 OR \u2018perineal\u2019) AND (\u2018hypospadia\u2019 OR \u2018anorectal malformations\u2019 OR \u2018duplication\u2019)\u201d.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Both patients presented were detected by physical examination. One case had a normal anus, while the other had anorectal malformations (ARMs). Both cases showed normal Karyotype (46XY). Magnetic resonance imaging (MRI) demonstrated a soft tissue lesion that has not connection with or communication with adjacent organs. Both of then underwent mass excision and urethroplasty after cystoscopy, which revealed a normal bladder and posterior urethra. Pathological result of perineal mass suggested the histological structures of urothelium with von Brunn's Nests. There were totally 8 cases of hypospadia with perineal mass reported in the literature. Reported histological findings included 3 (37.5%) colonic structure, 2 (25%) lipoma, 2 (25%) accessory scrotum with hamartom and 1 (12.5%) sacrococcygeal teratomain.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Discussion<\/jats:title>\n                    <jats:p>Perineal mass with proximal hypospadias is an exceptionally rare anomaly. The histopathological founding of perineal mass lined by urothelium with von Brunn's nests has not been previously reported.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.3389\/fped.2026.1876663","type":"journal-article","created":{"date-parts":[[2026,8,24]],"date-time":"2026-08-24T05:28:18Z","timestamp":1787549298000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Urothelial-lined perineal masses in hypospadias: clinical and pathological insights"],"prefix":"10.3389","volume":"14","author":[{"given":"Enfu","family":"Huang","sequence":"first","affiliation":[{"name":"Department of Urology, Children's Hospital of Soochow University","place":["Jiangsu, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chao","family":"Wang","sequence":"additional","affiliation":[{"name":"Department of Urology, Children's Hospital of Soochow University","place":["Jiangsu, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Li","family":"Tao","sequence":"additional","affiliation":[{"name":"Department of Urology, Children's Hospital of Soochow University","place":["Jiangsu, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jie","family":"Zhu","sequence":"additional","affiliation":[{"name":"Department of Surgery, Children's Hospital of Soochow University","place":["Jiangsu, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ting","family":"Zhang","sequence":"additional","affiliation":[{"name":"Department of Urology, Children's Hospital of Soochow University","place":["Jiangsu, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiangming","family":"Yan","sequence":"additional","affiliation":[{"name":"Department of Urology, Children's Hospital of Soochow University","place":["Jiangsu, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xueming","family":"Zhu","sequence":"additional","affiliation":[{"name":"Department of Pathology, Children's Hospital of Soochow University","place":["Jiangsu, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yun","family":"Zhou","sequence":"additional","affiliation":[{"name":"Department of Urology, Children's Hospital of Soochow University","place":["Jiangsu, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2026,8,24]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","first-page":"188","DOI":"10.1016\/j.jpedsurg.2004.09.027","article-title":"Surgical management of perineal masses in patients with anorectal malformations","volume":"40","author":"Shaul","year":"2005","journal-title":"J Pediatr Surg"},{"key":"B2","doi-asserted-by":"publisher","first-page":"1263","DOI":"10.1097\/01.ju.0000113425.79116.b6","article-title":"Ulaanbaatar procedure for tubularization of the glans in severe hypospadias","volume":"171","author":"Dewan","year":"2004","journal-title":"J Urol"},{"key":"B3","doi-asserted-by":"publisher","first-page":"175","DOI":"10.1016\/j.urology.2016.03.032","article-title":"A new modification of the Koyanagi technique for the one-stage repair of severe hypospadias","volume":"93","author":"Kang","year":"2016","journal-title":"Urology"},{"key":"B4","doi-asserted-by":"publisher","first-page":"532","DOI":"10.1007\/s00383-002-0785-4","article-title":"Mucosal-lined hemiperineal anomaly possibly caused by antenatal rupture of a caudal duplication","volume":"18","author":"Thomas","year":"2002","journal-title":"Pediatr Surg Int"},{"key":"B5","doi-asserted-by":"publisher","first-page":"478","DOI":"10.1007\/s00383-005-1416-7","article-title":"Perineal hamartoma with accessory scrotum, anorectal anomaly, and hypospadias-a rare association with embryological significance: case report","volume":"21","author":"Ramzisham","year":"2005","journal-title":"Pediatr Surg Int"},{"key":"B6","doi-asserted-by":"publisher","first-page":"979","DOI":"10.1007\/s00383-006-1800-y","article-title":"Perineal lipomas associated with anorectal malformations","volume":"22","author":"Wester","year":"2006","journal-title":"Pediatr Surg Int"},{"key":"B7","doi-asserted-by":"publisher","first-page":"e5","DOI":"10.1016\/j.jpedsurg.2011.03.083","article-title":"Perineal mass protrusion with rectal mucosa: a rectal duplication that underwent exstrophy","volume":"46","author":"Sun","year":"2011","journal-title":"J Pediatr Surg"},{"key":"B8","doi-asserted-by":"publisher","first-page":"200","DOI":"10.1016\/j.urology.2020.04.050","article-title":"Urethral duplication with heterotopic colonic epithelium presenting as penoscrotal mass: a rare association","volume":"142","author":"Danish","year":"2020","journal-title":"Urology"},{"key":"B9","doi-asserted-by":"publisher","first-page":"e71","DOI":"10.1055\/s-0040-1715182","article-title":"Colorectal hamartoma presenting as a perineal mass in a boy with proximal hypospadias","volume":"8","author":"Wolffenbuttel","year":"2020","journal-title":"European J Pediatr Surg Rep"},{"key":"B10","doi-asserted-by":"publisher","first-page":"627188","DOI":"10.3389\/fped.2021.627188","article-title":"Anorectal malformations associated with labioscrotal fold malformation and perineal mass in pediatric patients: over a decade of experience","volume":"9","author":"Wang","year":"2021","journal-title":"Front Pediatr"},{"key":"B11","doi-asserted-by":"publisher","first-page":"881","DOI":"10.1016\/j.radcr.2021.12.033","article-title":"Prenatally diagnosed accessory scrotum: a case report and review of the literature on prenatal features","volume":"17","author":"Deguchi","year":"2022","journal-title":"Radiol Case Rep"},{"key":"B12","doi-asserted-by":"publisher","first-page":"631","DOI":"10.1097\/JU.0000000000004715","article-title":"Long-term risk of reoperation after hypospadias surgery in childhood: a data-linkage study","volume":"214","author":"Vavilov","year":"2025","journal-title":"J Urol"},{"key":"B13","first-page":"501","article-title":"Accessory scrotum with perineal lipoma diagnosed prenatally: case report and review of the literature","volume":"77","author":"Murase","year":"2015","journal-title":"Nagoya J Med Sci"},{"key":"B14","doi-asserted-by":"publisher","first-page":"102","DOI":"10.1016\/j.jpag.2013.06.014","article-title":"Congenital perineal mass with cervovaginal duplication: a case report and literature review","volume":"27","author":"Liu","year":"2014","journal-title":"J Pediatr Adolesc Gynecol"},{"key":"B15","doi-asserted-by":"publisher","first-page":"733","DOI":"10.1046\/j.1464-410X.2002.02656.x","article-title":"The urethral plate\u2013does it grow into the genital tubercle or within it","volume":"89","author":"Penington","year":"2002","journal-title":"BJU Int"},{"key":"B16","doi-asserted-by":"publisher","first-page":"107","DOI":"10.3978\/j.issn.1000-9604.2016.02.01","article-title":"The pathology of urinary bladder lesions with an inverted growth pattern","volume":"28","author":"Guo","year":"2016","journal-title":"Chin J Cancer Res"},{"key":"B17","doi-asserted-by":"publisher","first-page":"317","DOI":"10.1016\/S0022-5347(17)56384-3","article-title":"The prevalence and significance of Brunn\u2019s nests, cystitis cystica and squamous metaplasia in normal bladders","volume":"122","author":"Wiener","year":"1979","journal-title":"J Urol"},{"key":"B18","doi-asserted-by":"publisher","first-page":"523","DOI":"10.1080\/15513815.2020.1854401","article-title":"Bladder exstrophy polyp: an uncommon entity in surgical pathology","volume":"41","author":"Bannoura","year":"2022","journal-title":"Fetal Pediatr Pathol"},{"key":"B19","doi-asserted-by":"publisher","first-page":"74","DOI":"10.1016\/j.diff.2018.08.001","article-title":"Development of the human penis and clitoris","volume":"103","author":"Baskin","year":"2018","journal-title":"Differentiation"},{"key":"B20","doi-asserted-by":"publisher","first-page":"86","DOI":"10.1016\/j.diff.2018.08.002","article-title":"Human glans and preputial development","volume":"103","author":"Liu","year":"2018","journal-title":"Differentiation"},{"key":"B21","doi-asserted-by":"publisher","first-page":"877","DOI":"10.1016\/S0022-3468(99)90391-3","article-title":"Septation and differentiation of the embryonic human cloaca","volume":"34","author":"Paidas","year":"1999","journal-title":"J Pediatr Surg"},{"key":"B22","doi-asserted-by":"publisher","first-page":"353","DOI":"10.1111\/j.1469-7580.2005.00459.x","article-title":"Cellular proliferation in the urorectal septation complex of the human embryo at Carnegie stages 13-18: a nuclear area-based morphometric analysis","volume":"207","author":"Nebot-Cegarra","year":"2005","journal-title":"J Anat"},{"key":"B23","doi-asserted-by":"publisher","first-page":"623","DOI":"10.1046\/j.1464-410x.2001.02136.x","article-title":"Neoplasia in adult exstrophy patients","volume":"87","author":"Smeulders","year":"2001","journal-title":"BJU Int"},{"key":"B24","doi-asserted-by":"publisher","first-page":"1522","DOI":"10.1097\/01.ju.0000179240.25781.1b","article-title":"Polyps in the exstrophic bladder. A cause for concern","volume":"174","author":"Novak","year":"2005","journal-title":"J Urol"},{"key":"B25","doi-asserted-by":"publisher","first-page":"162385","DOI":"10.1016\/j.jpedsurg.2025.162385","article-title":"Bulbospongiosus muscle reconstruction in proximal hypospadias: post pubertal function and patient satisfaction","volume":"60","author":"Hennayake","year":"2025","journal-title":"J Pediatr Surg"},{"key":"B26","doi-asserted-by":"publisher","first-page":"162074","DOI":"10.1016\/j.jpedsurg.2024.162074","article-title":"Repair of the bulbospongiosus muscle to suspend the penis to the pubic bones in proximal hypospadias","volume":"60","author":"Hennayake","year":"2025","journal-title":"J Pediatr Surg"}],"container-title":["Frontiers in Pediatrics"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fped.2026.1876663\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,24]],"date-time":"2026-08-24T05:28:20Z","timestamp":1787549300000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fped.2026.1876663\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,8,24]]},"references-count":26,"alternative-id":["10.3389\/fped.2026.1876663"],"URL":"https:\/\/doi.org\/10.3389\/fped.2026.1876663","relation":{},"ISSN":["2296-2360"],"issn-type":[{"value":"2296-2360","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,8,24]]},"article-number":"1876663"}}