Congenital prepubic sinus: A case report of local experience within a sub-Saharan setting

Congenital prepubic sinus: A case report and review of the literature

BackgroundCongenital 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 summaryA 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.ConclusionsBefore 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.

Congenital prepubic sinus is an extremely rare anomaly. The etiology is uncertain and the anatomical features often differ from each other. We report a 22-year-old woman with a congenital prepubic sinus accompanied by a prevesical abscess. She was admitted to our hospital with high-grade fever and low abdominal pain. Computed tomography revealed a prevesical abscess. After treatment of the prevesical abscess, we completely excised the congenital prepubic sinus. To our knowledge, this is the first reported case that accompanied by prevesical abscess on a congenital prepubic sinus. Moreover, this case represents the oldest reported age of a patient with a congenital prepubic sinus.

Congenital prepubic sinus is a rare disorder with fewer than 30 cases reported in the literature. The origin of the sinus and its tract is unclear but urethral duplication, cloacal remnant or midline closure defect are amongst the postulated mechanisms for the development of the sinus. Chronic discharge and infection are the main indications for surgery. Although surgery is the definite way of correction, timing and extend of surgery are not well documented due to the limited number of cases. Here we present two cases with congenital prepubic sinus, a four month old girl and a five years old boy, who were admitted with discharge through the external opening of the prepubic sinus which are shown not to have a connection with urinary system and are managed by limited surgical excision. Postoperative courses were uneventful and no relapse or complications were encountered. Histological examination revealed that the sinus tract is lined with squamous stratified epithelium without any other significant finding. Although the classification and definition of congenital prepubic sinus is not clear, extensive imaging modalities and surgical procedures are not necessary in most conditions as these can create additional burden for the patient and the healthcare system and may cause complications at the cost of nothing.

Congenital prepubic sinus is a rare anomaly: only five pediatric cases have to our knowledge been reported to date. We report two further cases with the sinus extending from the overlying skin through the rectus fascia to the anterior bladder wall. In our first case the opening of the sinus was just 3 cm below the midline inferior to the navel, with concentric, thin smooth-muscle layers. This lesion may represent a mild forme fruste of a midline closure defect. In our second case, the sinus was lined by transitional epithelium in its distal portion with stratified squamous epithelium near the surface, and was also surrounded by a thin smooth-muscle layer. These two cases may represent a variant of epispadiac urethral duplication. Our observations suggest that congenital prepubic sinus and urethral duplication are related embryologically.

CONGENITAL PREPUBIC SINUS: A VARIANT OF DORSAL URETHRAL DUPLICATION SUGGESTED BY IMMUNOHISTOCHEMICAL ANALYSIS

Congenital prepubic sinus: Rare diagnosis with unclear etiology

Congenital prepubic sinus is an exceptional disorder of uncertain origin. These sinuses may represent a variant of epispadiac duplication or dorsal urethral duplication of the urethra.We present a case of isolated blind ending prepubic congenital sinus in a 5-year-old male child, which revealed transitional epithelium lining proximally, and squamous epithelium distally. This entity presents with either an opening in the prepubic region and/or persistent mucoid discharge. Excision leads to a permanent cure. Keywords :Congenital prepubic sinus, Urethral duplication, Epispadias. African Journal of Paediatric Surgery Vol. 4 (1) 2007: pp. 49-50

Introduction: Congenital prepubic sinus (CPS) is a rare congenital abnormality in the urinary tract in the form of remnant ducts that extend from skin openings near the pubic symphysis to various areas. Due to its anatomical and pathological variation, the clinical manifestation of this condition has a wide spectrum. This paper shows one of the youngest reported cases of CPS that was successfully treated with surgery. This paper aims to elucidate and provide the current literature review in order to increase awareness towards CPS diagnosis in general practitioners, pediatricians, and surgeons. Case report: Here we present a 2-months-old female baby with a CPS that presented a purulent discharge and recurrent abscess from an opening since birth. Prior to surgery, the patient had visited multiple health care provider until a definitive diagnosis of CPS could be established, which was followed by the administration of topical antibiotic prior to surgery. The condition was diagnosed through abdominal CT imaging and treated with a simple surgical technique aided with methylene blue dye. Conclusion: Clinicians should consider the differential diagnosis of congenital anomaly such as CPS in a patient presenting with purulent discharge or erythema from accessory meatus in the pubic region.

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.

Congenital prepubic sinus (CPS) is a rare diagnosis. It is defined as a blind-ending tract originating from the midline of the genital region. There are three types of CPS classified according to the course of the tract and location of the skin opening. The etiology is thought to be an intussusception during fusion of the abdominal wall or, alternatively, incomplete urethral duplication. We report on a two-year-old boy with a skin fistula on the dorsal side of the penis. A slight secretion occurred when the surrounding subcutaneous tissue was compressed. After total resection of the sinus, histological examination revealed that the tract was lined primarily with multilayered epithelium. Immunohistochemical studies showed that the sinus was lined with transitional and squamous epithelium. At the base the lining epithelium was transitional and shifted distally to noncornifying squamous epithelium. The epithelial layer therefore corresponded to the inner surface of the urethra, thus supporting the assumption that CPS results from incomplete urethral duplication. The immunohistochemical examination of the epithelium of Type II CPS proved, in this case, the existence of urothelium as the inner surface of the sinus. In view of this evidence it appears likely that the congenital prepubic sinus can be classified etiologically as an incomplete urethral duplication.

Congenital prepubic sinus (CPS) is a very rare congenital anomaly. The baby generally present with discharge from an opening situated in midline below umbilicus to root of penis in male or clitoris in female child. The etiology of this congenital anomaly is not exactly known. There is debate in etiology as one theory support anomalous anterior abdominal wall closure and another support variant of dorsal urethral duplication. Here we are presenting the six cases of CPS with their management and brief discussion on theories that have been proposed for their etiology.

Congenital prepubic sinus is a rare congenital anomaly situated in the midline of the lower abdomen. We report a case of congenital prepubic sinus, closely associated with a urachal remnant. Preoperative magnetic resonance imaging showed clearly that the sinus tracked the urachus caudally. This finding supports the theory that the anomaly is caused by abnormal remnant tissue originating from the cloacal membrane, which tracks the allantois duct caudally along with fetal longitudinal growth.

Congenital prepubic sinus: an epispadiac variant of urethral duplication: Case report and review of literature

We report two cases of congenital prepubic sinus (CPS) and discuss theories about its embryology and the etiology of its variants to improve the global understanding of this uncommon anomaly. Based on a review of the scant number of reported cases and our own experience, we postulate that CPS may be caused by a residual cloacal membrane and umbilicophallic groove and that its depth may determine the position of its ending.