{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,5]],"date-time":"2026-09-05T17:11:03Z","timestamp":1788628263271,"version":"build-2803163510"},"reference-count":57,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2024,6,11]],"date-time":"2024-06-11T00:00:00Z","timestamp":1718064000000},"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><jats:title>Introduction<\/jats:title><jats:p>Urethral multiplicity is a rare congenital anomaly characterized by the presence of two or more urethral channels. It is more common in males and can cause double urinary stream, incontinence, obstruction, and recurrent urinary infections. Diagnosis is difficult due to diverse clinical manifestations. Implementing an evidence-based treatment plan is challenging due to the need for more concise and informative summary publications. Our paper provides a comprehensive review of the management of this pathology and might serve as a valuable resource for pediatric urologists and specialists in the field.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>A comprehensive search in four electronic databases, PubMed\u00ae, PubMed Central\u00ae (PMC), Scopus, and Clarivate Analytics's Web of Science (WoS), was conducted to identify case reports and series published between 2008 and 2023 on urethral multiplicities. The quality of the articles was assessed using qualified instruments. Covidence\u00ae tool-guided synthesis was followed by individual patient data extraction. Further classifications and analysis were made using Microsoft Excel\u00ae.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Out of the 90 papers included in the review, 62 were case presentations, and 28 were case series. We found 250 boys with urethral multiplicity. Based on Effman's classification, there were 38 cases of type I (15.3%), 21 type IIA1 (8.4%), 55 type IIA2 (22.1%), 91 type IIA2Y (36.5%), 4 type IIB (1.6%), and 6 type III (2.4%) urethral duplications. There were 19 cases of prepubic sinuses (7.6%), 9 triplications (3.6%), and 6 unknown forms (2.4%). We have provided data for each type, including clinical presentation, investigations, surgical management, and outcomes.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Urethral multiplicities are a rare and varied group of malformations that require high-quality imaging examination for successful management. Treatment is specific to each patient and may depend on the surgeon's preference or skill.<\/jats:p><\/jats:sec><jats:sec><jats:title>Systematic Review Registration<\/jats:title><jats:p><jats:ext-link>https:\/\/www.crd.york.ac.uk\/prospero\/display_record.php?ID\u2009=\u2009CRD42023471685<\/jats:ext-link>, identifier (CRD42023471685).<\/jats:p><\/jats:sec>","DOI":"10.3389\/fped.2024.1404947","type":"journal-article","created":{"date-parts":[[2024,6,12]],"date-time":"2024-06-12T13:33:57Z","timestamp":1718199237000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":7,"title":["Urethral multiplicity in boys: systematic review of case reports and case series from the last 15 years"],"prefix":"10.3389","volume":"12","author":[{"given":"Horea","family":"Gozar","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zsolt","family":"Bara","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Evelyn","family":"Kovacs","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Iulia","family":"Gozar","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zoltan","family":"Derzsi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2024,6,11]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","first-page":"216","DOI":"10.1016\/j.urology.2020.06.074","article-title":"Urethral multiplicity: when two are better than one","volume":"146","author":"Janssen","year":"2020","journal-title":"Urology"},{"key":"B2","doi-asserted-by":"publisher","first-page":"1064","DOI":"10.1016\/j.jpurol.2013.03.006","article-title":"Urethral duplication\u2013a wide spectrum of anomalies","volume":"9","author":"Onofre","year":"2013","journal-title":"J Pediatr Urol"},{"key":"B3","doi-asserted-by":"publisher","first-page":"156","DOI":"10.4103\/0970-1591.174780","article-title":"Urethral duplication with unusual cause of bladder outlet obstruction","volume":"32","author":"Venkatramani","year":"2016","journal-title":"Indian J Urol"},{"key":"B4","doi-asserted-by":"publisher","first-page":"179","DOI":"10.1148\/119.1.179","article-title":"Duplication of the urethra","volume":"119","author":"Effmann","year":"1976","journal-title":"Radiology"},{"key":"B5","doi-asserted-by":"publisher","first-page":"e1","DOI":"10.1016\/j.jpurol.2016.10.008","article-title":"Practical and functional classification of the double urethra: a variable, complex and fascinating malformation observed in 20 patients","volume":"13","author":"Lima","year":"2017","journal-title":"J Pediatr Urol"},{"key":"B6","doi-asserted-by":"publisher","first-page":"138","DOI":"10.4103\/jiaps.JIAPS_62_18","article-title":"Hypospadiac urethral duplication-a rare variant and novel use of snodgrass urethroplasty","volume":"24","author":"Saxena","year":"2019","journal-title":"J Indian Assoc Pediatr Surg"},{"key":"B7","doi-asserted-by":"publisher","first-page":"52","DOI":"10.2478\/amma-2013-0012","article-title":"Two cases of dorsal urethral duplication","volume":"59","author":"Gozar","year":"2013","journal-title":"Acta Med Marisiensis"},{"key":"B8","doi-asserted-by":"publisher","first-page":"60","DOI":"10.1136\/BMJEBM-2017-110853","article-title":"Methodological quality and synthesis of case series and case reports","volume":"23","author":"Murad","year":"2018","journal-title":"BMJ Evid Based Med"},{"key":"B9","doi-asserted-by":"publisher","first-page":"2127","DOI":"10.11124\/JBISRIR-D-19-00099","article-title":"Methodological quality of case series studies: an introduction to the JBI critical appraisal tool","volume":"18","author":"Munn","year":"2020","journal-title":"JBI Evid Synth"},{"key":"B10","doi-asserted-by":"publisher","first-page":"163","DOI":"10.1097\/XEB.0000000000000064","article-title":"Conducting systematic reviews of association (etiology): the Joanna Briggs Institute\u2019s approach","volume":"13","author":"Moola","year":"2015","journal-title":"Int J Evid Based Healthc"},{"key":"B11","doi-asserted-by":"publisher","first-page":"119","DOI":"10.1097\/01.XPS.0000469366.65557.5e","article-title":"A rare case of urethral triplication in association with tethered cord and vertebral anomalies","volume":"12","author":"Kurian","year":"2016","journal-title":"Ann Pediatr Surg"},{"key":"B12","doi-asserted-by":"publisher","first-page":"198","DOI":"10.1016\/j.urology.2020.05.053","article-title":"Urethral triplication with diverticulum malformation: a case report and literature review","volume":"144","author":"Zhang","year":"2020","journal-title":"Urology"},{"key":"B13","doi-asserted-by":"publisher","first-page":"186","DOI":"10.1016\/j.urology.2010.09.019","article-title":"Urethral triplication and urethrovasal reflux in 5-day-old male infant","volume":"78","author":"Kajbafzadeh","year":"2011","journal-title":"Urology"},{"key":"B14","doi-asserted-by":"publisher","first-page":"1227","DOI":"10.1007\/s00247-009-1345-z","article-title":"Urethral triplication with meatuses terminating on the penis: a rare case presenting with bifid urinary stream","volume":"39","author":"Hirselj","year":"2009","journal-title":"Pediatr Radiol"},{"key":"B15","doi-asserted-by":"publisher","first-page":"29","DOI":"10.1016\/j.eucr.2018.11.018","article-title":"Urethral duplication in a child with VATER association","volume":"23","author":"Nerli","year":"2019","journal-title":"Urol Case Rep"},{"key":"B16","doi-asserted-by":"publisher","first-page":"16","DOI":"10.1016\/j.epsc.2016.04.001","article-title":"Congenital prepubic sinus: rare diagnosis with unclear etiology","volume":"9","author":"Nonnenmacher","year":"2016","journal-title":"J Pediatr Surg Case Rep"},{"key":"B17","doi-asserted-by":"publisher","first-page":"102749","DOI":"10.1016\/j.epsc.2023.102749","article-title":"Urethral duplication presenting as a prepubic sinus in a male: a case report","volume":"100","author":"Nascimben","year":"2024","journal-title":"J Pediatr Surg Case Rep"},{"key":"B18","doi-asserted-by":"publisher","first-page":"e82","DOI":"10.1016\/j.jpurol.2012.09.010","article-title":"Congenital prepubic sinus: a variant of epispadiac dorsal urethral duplication","volume":"9","author":"Nasir","year":"2013","journal-title":"J Pediatr Urol"},{"key":"B19","doi-asserted-by":"publisher","first-page":"36","DOI":"10.1016\/j.epsc.2016.12.003","article-title":"Congenital pre-pubic sinus: an embryological enigma","volume":"18","author":"Sinha","year":"2017","journal-title":"J Pediatr Surg Case Rep"},{"key":"B20","doi-asserted-by":"publisher","first-page":"196","DOI":"10.1016\/j.afju.2015.12.001","article-title":"Congenital prepubic sinus\u2014a variant of urethral duplication","volume":"22","author":"Aihole","year":"2016","journal-title":"Afr J Urol"},{"key":"B21","doi-asserted-by":"publisher","first-page":"881","DOI":"10.4111\/kju.2007.48.8.881","article-title":"Congenital prepubic sinus: a variant of urethral duplication","volume":"48","author":"Kim","year":"2007","journal-title":"Korean J Urol"},{"key":"B22","doi-asserted-by":"publisher","first-page":"165","DOI":"10.1186\/s40792-021-01245-0","article-title":"A totally extraperitoneal approach for endoscopic resection of a congenital prepubic sinus through the pubic symphysis","volume":"7","author":"Fukuhara","year":"2021","journal-title":"Surg Case Rep"},{"key":"B23","doi-asserted-by":"publisher","first-page":"205","DOI":"10.1016\/j.urology.2022.09.024","article-title":"Congenital anterior urethral diverticulum in identical twins","volume":"171","author":"Wei","year":"2023","journal-title":"Urology"},{"key":"B24","doi-asserted-by":"publisher","first-page":"PJ1","DOI":"10.7860\/JCDR\/2017\/30269.10665","article-title":"A boy with a congenital sagittal fissure of the glans penis representing an abortive isolated urethral duplication","volume":"11","author":"Sfoungaris","year":"2017","journal-title":"J Clin Diagn Res"},{"key":"B25","doi-asserted-by":"publisher","first-page":"451","DOI":"10.1016\/j.urology.2013.02.010","article-title":"A new variation of urethral duplication","volume":"82","author":"Sencan","year":"2013","journal-title":"Urology"},{"key":"B26","doi-asserted-by":"publisher","first-page":"76","DOI":"10.1177\/1756287214561760","article-title":"The double urethra: revisiting the surgical classification","volume":"7","author":"Abouzeid","year":"2015","journal-title":"Ther Adv Urol"},{"key":"B27","doi-asserted-by":"publisher","first-page":"62","DOI":"10.1007\/s12262-016-1472-3","article-title":"Hypospadiac duplication of anterior urethra\u2014a rare congenital anomaly","volume":"79","author":"Goyal","year":"2017","journal-title":"Indian J Surg"},{"key":"B28","doi-asserted-by":"publisher","first-page":"2216","DOI":"10.1016\/j.jpedsurg.2019.12.012","article-title":"Urethral duplication in male children: a study of 12 cases","volume":"55","author":"Kang","year":"2020","journal-title":"J Pediatr Surg"},{"key":"B29","doi-asserted-by":"publisher","first-page":"4707","DOI":"10.22038\/ijp.2017.22681.1895","article-title":"Novel presentation of complete coronal urethral duplication: a case report","volume":"5","author":"Salimi","year":"2017","journal-title":"Int J Pediatr"},{"key":"B30","doi-asserted-by":"publisher","first-page":"e3620","DOI":"10.5812\/ijp.3620","article-title":"Complete urethral duplication in children: a case report","volume":"26","author":"Roshanzamir","year":"2016","journal-title":"Iran J Pediatr"},{"key":"B31","doi-asserted-by":"publisher","first-page":"601","DOI":"10.1007\/s11255-013-0424-0","article-title":"An unusual variant of urethral duplication: an addition to the Effman classification","volume":"45","author":"Abraham","year":"2013","journal-title":"Int Urol Nephrol"},{"key":"B32","doi-asserted-by":"publisher","first-page":"313","DOI":"10.4103\/0189-6725.91667","article-title":"Urethral duplication in a 12-year-old child","volume":"8","author":"Licien Mouafo Tambo","year":"2011","journal-title":"Afr J Paediatr Surg"},{"key":"B33","doi-asserted-by":"publisher","first-page":"572","DOI":"10.4274\/balkanmedj.2017.0471","article-title":"The association of congenital urethral duplication and double megalourethra","volume":"34","author":"U\u00e7ar","year":"2017","journal-title":"Balkan Med J"},{"key":"B34","doi-asserted-by":"publisher","first-page":"99","DOI":"10.1016\/j.afju.2013.01.003","article-title":"Bladder exstrophy associated with complete urethral duplication: bladder can be augmented with dorsal urethral mucosa","volume":"19","author":"Mensah","year":"2013","journal-title":"Afr J Urol"},{"key":"B35","doi-asserted-by":"publisher","first-page":"275","DOI":"10.4103\/jiaps.JIAPS_164_18","article-title":"Urethral duplication in children: experience of twenty cases","volume":"24","author":"Wani","year":"2019","journal-title":"J Indian Assoc Pediatr Surg"},{"key":"B36","doi-asserted-by":"publisher","first-page":"229","DOI":"10.23736\/S0393-2249.19.03271-5","article-title":"Urethral duplication in male epispadias: a very uncommon association","volume":"72","author":"Caione","year":"2020","journal-title":"Minerva Urol Nefrol"},{"key":"B37","doi-asserted-by":"publisher","first-page":"188","DOI":"10.1016\/j.jpurol.2009.07.006","article-title":"Urethral duplication: cases of ventral and dorsal complete duplication and review of the literature","volume":"6","author":"Coleman","year":"2010","journal-title":"J Pediatr Urol"},{"key":"B38","doi-asserted-by":"publisher","first-page":"15","DOI":"10.2147\/RRU.S239106","article-title":"Complete urethral duplication: case report and literature review","volume":"12","author":"Suoub","year":"2020","journal-title":"Res Rep Urol"},{"key":"B39","doi-asserted-by":"publisher","first-page":"343","DOI":"10.1016\/j.jpurol.2011.06.011","article-title":"Complete urethral duplication: description of surgical approach mimicking distal epispadias repair","volume":"8","author":"Alanee","year":"2012","journal-title":"J Pediatr Urol"},{"key":"B40","doi-asserted-by":"publisher","first-page":"1144","DOI":"10.1590\/S1677-5538.IBJU.2016.0269","article-title":"Urethral duplication type influences on the complications rate and number of surgical procedures","volume":"43","author":"Lopes","year":"2017","journal-title":"Int Braz J Urol"},{"key":"B41","doi-asserted-by":"publisher","first-page":"903","DOI":"10.1016\/j.urology.2008.12.040","article-title":"Complex case of urethral duplication with megalourethra","volume":"74","author":"Sindjic","year":"2009","journal-title":"Urology"},{"key":"B42","doi-asserted-by":"publisher","first-page":"20","DOI":"10.12688\/f1000research.3848.1","article-title":"Congenital duplication of urethra with urethral diverticulum: a rare case report","volume":"114","author":"Shah","year":"2014","journal-title":"BJU Int"},{"key":"B43","doi-asserted-by":"publisher","first-page":"115","DOI":"10.1007\/s00383-010-2720-4","article-title":"Minimally invasive repair of hypospadiac urethral duplication","volume":"27","author":"Yanai","year":"2011","journal-title":"Pediatr Surg Int"},{"key":"B44","doi-asserted-by":"publisher","first-page":"1729","DOI":"10.1007\/s11255-014-0740-z","article-title":"Multiple hypoplastic duplicated urethral hydrodistension and simple end-to-end anastomosis to penoscrotal hypospadias: a novel technique","volume":"46","author":"Mazaheri","year":"2014","journal-title":"Int Urol Nephrol"},{"key":"B45","doi-asserted-by":"publisher","first-page":"1487","DOI":"10.1007\/s00383-022-05186-z","article-title":"Anorectal malformation, urethral duplication, occult spinal dysraphism (ARM-UD-OSD): a challenging uncommon association","volume":"38","author":"Lena","year":"2022","journal-title":"Pediatr Surg Int"},{"key":"B46","doi-asserted-by":"publisher","first-page":"1335","DOI":"10.1016\/j.jpedsurg.2016.11.034","article-title":"Fate of males with urethral \u201cY-duplication\u201d: 40-year long follow-up in 8 patients","volume":"52","author":"Lima","year":"2017","journal-title":"J Pediatr Surg"},{"key":"B47","doi-asserted-by":"publisher","first-page":"245","DOI":"10.1055\/s-0035-1551569","article-title":"Y-type urethral duplication: a true variant of the anomaly or a misnomer?","volume":"26","author":"AbouZeid","year":"2016","journal-title":"Eur J Pediatr Surg"},{"key":"B48","doi-asserted-by":"publisher","first-page":"95","DOI":"10.1016\/0022-3468(77)90302-5","article-title":"\u201cH-type\u201d urethroanal fistula","volume":"12","author":"Stephens","year":"1977","journal-title":"J Pediatr Surg"},{"key":"B49","doi-asserted-by":"publisher","first-page":"501","DOI":"10.1148\/radiology.194.2.7824732","article-title":"Congenital urethroperineal fistula","volume":"194","author":"Bates","year":"1995","journal-title":"Radiology"},{"key":"B50","doi-asserted-by":"publisher","first-page":"414.e1","DOI":"10.1016\/j.jpurol.2021.02.008","article-title":"Working classification and the quest for an effective, reconstructive management strategy in Y duplication of male urethra","volume":"17","author":"Kurian","year":"2021","journal-title":"J Pediatr Urol"},{"key":"B51","doi-asserted-by":"publisher","first-page":"2068","DOI":"10.1016\/j.juro.2015.01.082","article-title":"Q-island flap urethroplasty: 1-stage procedure for reconstruction of Y-type urethral duplications in children","volume":"193","author":"Raffoul","year":"2015","journal-title":"J Urol"},{"key":"B52","doi-asserted-by":"publisher","first-page":"613.e1","DOI":"10.1016\/j.jpurol.2017.04.011","article-title":"The anterior sagittal transrectal approach (ASTRA) for cases associated with rectal implantation of the urethra: a retrospective review of six cases","volume":"13","author":"Macedo","year":"2017","journal-title":"J Pediatr Urol"},{"key":"B53","doi-asserted-by":"publisher","first-page":"363","DOI":"10.1016\/j.jpurol.2009.01.006","article-title":"Urethral duplication in children: our experience of eight cases","volume":"5","author":"Mane","year":"2009","journal-title":"J Pediatr Urol"},{"key":"B54","doi-asserted-by":"publisher","first-page":"385.e1","DOI":"10.1016\/j.jpurol.2020.02.010","article-title":"Urethral duplication-epidemiology, diagnosis, and treatment in a case series of 19 patients","volume":"16","author":"Guglielmetti","year":"2020","journal-title":"J Pediatr Urol"},{"key":"B55","doi-asserted-by":"publisher","first-page":"255","DOI":"10.1016\/j.jpurol.2008.02.001","article-title":"Complete duplication of bladder and urethra in a sagittal plane in a male infant: case report and literature review","volume":"4","author":"Coker","year":"2008","journal-title":"J Pediatr Urol"},{"key":"B56","doi-asserted-by":"publisher","first-page":"214","DOI":"10.1016\/j.urology.2012.03.031","article-title":"Treatment and medical follow-up of a boy with urethral triplication","volume":"80","author":"Lin","year":"2012","journal-title":"Urology"},{"key":"B57","doi-asserted-by":"publisher","first-page":"98","DOI":"10.1016\/j.jpurol.2014.11.009","article-title":"Combined robotic and open approach to excision of accessory bladder and urethral triplication","volume":"11","author":"Bowen","year":"2015","journal-title":"J Pediatr Urol"}],"container-title":["Frontiers in Pediatrics"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fped.2024.1404947\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,6,12]],"date-time":"2024-06-12T13:34:02Z","timestamp":1718199242000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fped.2024.1404947\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,6,11]]},"references-count":57,"alternative-id":["10.3389\/fped.2024.1404947"],"URL":"https:\/\/doi.org\/10.3389\/fped.2024.1404947","relation":{},"ISSN":["2296-2360"],"issn-type":[{"value":"2296-2360","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,6,11]]},"article-number":"1404947"}}