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IntroductionUrethral 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.MethodsA comprehensive search in four electronic databases, PubMed®, PubMed Central® (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® tool-guided synthesis was followed by individual patient data extraction. Further classifications and analysis were made using Microsoft Excel®.ResultsOut 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.ConclusionsUrethral 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.Systematic Review Registrationhttps://www.crd.york.ac.uk/prospero/display_record.php?ID = CRD42023471685, identifier (CRD42023471685).

Introduction: Congenital prepubic sinuses are rare congenital anomalies of the urogenital tract with a varied presentation, but pain is an uncommon presenting symptom. Case presentation: This report describes a 14-year-old male who presented to the emergency department following five days of intermittent pain associated with a small opening superior to the base of the dorsum of the penis and superficial to the pubic symphysis. Surgical excision of the sinus tract revealed the sinus coursed towards the pubis and did not connect to the bladder. The histology of the tract revealed transitional epithelium surrounded by smooth muscle. Conclusion: Management of congenital prepubic sinuses relies upon prompt recognition, diagnosis, and surgical intervention to prevent complications.

Abstract Background Congenital prepubic sinus is a rare anomaly found in the midline of the lower abdomen. Congenital prepubic sinus is usually asymptomatic in neonates, and a diagnosis is often achieved later in life after spontaneous extrusion of purulent material from the pre-existing hole in the pubic region. We present a case of congenital prepubic sinus presenting with purulent discharge after circumcision. Case summary A 4-year-old Caucasian boy presented to our urology out-patient clinic with purulent discharge from the distal part of the dorsum of his penis. He had a history of circumcision performed at a different center, 6 months ago. His parents stated that although various antibiotics were used, the purulent discharge continued for 6 months and the child had no complaints before circumcision. His condition was reported as superficial dorsal venous thrombosis, known as penile Mondor disease, in magnetic resonance imaging that was performed in the previous hospital. A physical examination revealed a small pinhole lesion at the distal part of his penis and a rigid cylindrical tube extending to the proximal side of his penis. We performed fistulography by injecting contrast material through a small angiocatheter and confirmed the diagnosis of prepubic sinus. Surgical exploration was performed and a long sinus, apparently ending as a fibrous tract at the anterior surface of his pubic symphysis, was found and resected. Conclusions Before congenital prepubic sinus surgery, it is critically important to rule out penile Mondor disease and the possibility of a circumcision complication (especially infective complications) mimicking congenital prepubic sinus.

The congenital prepubic sinus is a rare type of fistula with a broad range of anatomical and histological diversity. We report the case of an 8-month-old boy with a congenital prepubic sinus where immunohistochemistry studies showed only squamous epithelium and no transitional epithelium. The etiology of this disorder is still unclear and the most recent case reports suggest that the fistula might be a variation of a dorsal urethral duplication. We recommend performing immunohistochemical studies to further clarify the etiology, as the fistula may be caused by different developmental disorders.