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.