Abstract: The prostatic utricle represents the caudal end of fused Mullerian ducts. An untreated prostatic utricle pouch with urethra-ejaculatory duct reflux can lead to recurrent epididymal orchitis and other complications. Recurrent urinary tract infections (UTI) may manifest in patients when it is associated with the posterior urethral valve. A 13-year-old boy presented with recurrent urinary tract infections complicated by an anterior urethral-cutaneous fistula. On imaging, he was found to have the posterior urethral valve, prostatic utricle cyst, and urethra-ejaculatory duct reflux. To the best of our knowledge, this is the first case report to describe the combination of these three findings in the same patient. Prostatic utricle cyst is a rare condition that should be considered in the differential of midline cysts of the lower male genitourinary tract. MCU/RGU should be performed in such cases to exclude urethral-ejaculatory duct reflux which may predispose to epididymal-orchitis. Early management of reflux by correcting the underlying anomaly will help prevent complications.