The prostatic utricle cyst is usually an asymptomatic enlargement of the persistent remnant of Mullerian duct in males, especially seen associated with hypospadias and usually needs no active intervention. However occasionally they can become symptomatic and can present with complaints of impotence, ejaculatory pain, difficult voiding, urinary retention and abdominal pain. Hematuria is a rare symptom in such patients and is usually due to infection. However rarely it can be a harbinger of a more sinister complication like a malignancy. Herein, we present an adolescent boy with hematuria due to malignant degeneration in a long-standing prostatic utricle cyst.
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