Congenital prepubic sinus: Rare diagnosis with unclear etiology

Nonnenmacher, Gabriel; Rhein, Dieter; Loff, Steffan

doi:10.1016/j.epsc.2016.04.001

Cited by 3 publications

(3 citation statements)

References 5 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Recent theories suggest that prepubic sinuses may be a form of urethral duplication despite different etiological concerns. Immunohistochemical studies showed the presence of transitional epithelial cells in prepubic sinuses ( 16 ). Our findings also support this suggestion: in almost all cases, the excised specimens showed the presence of urethral tissue.…”

Section: Discussionmentioning

confidence: 99%

Urethral multiplicity in boys: systematic review of case reports and case series from the last 15 years

Gozar,

Bara,

Kovacs

et al. 2024

Front. Pediatr.

View full text Add to dashboard Cite

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).

show abstract

Section: Discussionmentioning

confidence: 99%

Urethral multiplicity in boys: systematic review of case reports and case series from the last 15 years

Gozar,

Bara,

Kovacs

et al. 2024

Front. Pediatr.

View full text Add to dashboard Cite

show abstract

“…It is also reported in literature as suprapubic fistula, suprapubic dermoid sinus etc. After the first report by Campbell et al, in 1987, around 50 cases are reported in literature [1][2][3][4][5]. Four theories are proposed to explain the etiology of CPS: 1) anomalous abdominal wall closure; 2) a variant of dorsal urethral duplication; 3) a fistula of primitive urogenital sinus and 4) a remnant of primitive cloaca [6][7][8][9].…”

Section: Discussionmentioning

confidence: 99%

Congenital prepubic sinus—An aborted dorsal urethral duplication or a cloacal remnant?

Nazir

Khan

Qamar

2019

Journal of Pediatric Surgery

View full text Add to dashboard Cite

show abstract

“…3, 6 However, there are working groups that in immunohistochemical characterization have only observed the presence of squamous and non-transitional epithelium. 7…”

Section: Introductionmentioning

confidence: 99%

Congenital Prepubic Sinus as a Variant of Incomplete Urethral Dorsal Duplication: A Case Report with New Insights into its Immunohistochemical Characterization and a Comprehensive Literature Review

Montero

Anaut²,

González

et al. 2022

Int J Surg Pathol

View full text Add to dashboard Cite

Congenital prepubic sinus (PS) is an extremely infrequent malformation consisting of a prepubic fistulous tract that classically does not communicate with the genitourinary system. Previous studies centered on its immunohistochemical characterization have shown inconsistent results, and the etiology has not been clarified. We present the case of a 2-year-old male who presented since birth with a fistulous orifice on the dorsum of the penis. He had no associated symptoms. Under general anesthesia, the fistulous tract was explored, and methylene blue was instilled through it. After cystoscopically verifying the absence of communication with the urethra, a complete resection of the lesion was performed. The immunohistochemical study showed positivity for low and high molecular weight keratins and a transitional pattern for keratin 7 and GATA3, with positivity at cul de sac level and negativity at proximal level. These findings suggest that this lesion is an incomplete dorsal duplication variant.

show abstract

Congenital prepubic sinus: Rare diagnosis with unclear etiology

Cited by 3 publications

References 5 publications

Urethral multiplicity in boys: systematic review of case reports and case series from the last 15 years

Urethral multiplicity in boys: systematic review of case reports and case series from the last 15 years

Congenital prepubic sinus—An aborted dorsal urethral duplication or a cloacal remnant?

Congenital Prepubic Sinus as a Variant of Incomplete Urethral Dorsal Duplication: A Case Report with New Insights into its Immunohistochemical Characterization and a Comprehensive Literature Review

Contact Info

Product

Resources

About