Congenital prepubic sinus (CPS) is an extremely rare congenital anomaly of the external genitalia of uncertain etiology. Herein, a case of CPS, where immunohistochemical staining was performed to elucidate the etiology of the sinus, is reported. The results of the immunohistochemical study demonstrated the lining epithelium was transitional proximally and squamous distally. Congenital prepubic sinus (CPS) is a rare congenital anomaly which is associated with discharge from an opening overlying the pubis symphysis. Various names including congenital prepubic sinus, subpubic fistula, prepubic dermoid sinus, and suprapubic dermoid sinus, were used to identify these lesions.The etiology of CPS is still controversial. There are 2 theories that have been proposed, including an anomalous anterior abdominal wall closure or variant of dorsal urethral duplication. [1][2][3][4] To explore the etiology, we performed immunohistochemical study with 3 cytokeratin antibodies that are known to be specific for differentiation between transitional and squamous epithelium.
CASE REPORTA 3-year-old boy presented with a discharging sinus in the prepubic region since birth. A tiny opening was located in the midline prepubic area on the dorsal radix of the penis (Fig. 1A).He had no other signs and symptoms related to the genitourinary tract. Clinical and laboratory findings were normal.Ultrasonography found a linear tubular structure in the midline prepubic region. Fistulography showed a noncommunicating fistulous tract toward the bladder neck. Surgical exploration was carried out and found a 4 cm long tract that ended blindly as a fibrous band at the anterior surface of the pubic symphysis, which was then excised (Fig. 1B). The specimen was divided into 2 segments from the middle area, and the cross sections were embedded into 2 paraffin blocks labeled as proximal and distal parts, respectively. Routine hematoxylin and eosin stain was performed on the histological sections. Successive sections were prepared for immunohistochemical analysis with monoclonal antibodies (DAKO A/S Glostrup, Denmark), including low and high molecular weight cytokeratin (CK 7, 8 and 903).Hematoxylin and eosin stain demonstrated transitional and squamous epithelium lining the sinus ( Fig. 2A, B). Immunohistochemical study showed that the stains with 3 cytokeratin antibodies were moderately to strongly positive at the proximal part ( Fig. 2C). At the distal part, high molecular weight cytokeratin stain with CK903 antibody was still strongly positive ( Fig. 2D), while low molecular weight cytokeratin stain was weakly to moderately positive. These results suggested that the lining epithelium was transitional proximally and squamous distally.