A 5-year-old male presented with the history of whitish discharge from a midline sinus opening just above the pubis for 2 months. Attempted radiography of the sinus revealed a blind fistula and voiding cystourethrography was normal. The fistula was excised deep to the subpubic space without any evidence of connection to the lower urinary tract. Pathologic evaluation of the lesion revealed a ciliated-columnar lining with stratified-squamous and transitional epithelium. To our knowledge, a subpubic sinus with this unique presentation of epithelium has not been reported previously.