A parameatal urethral cyst, which is a very rare congenital anomaly, was first reported in two male cases in 1956 by Thompson and Lantin. We report the case of a 20-year-old male having a spherical, cystic swelling, 7 mm in size at the external urethral meatus. The diagnosis was made by physical examination and ultrasonography. The cyst was completely excised under general anesthesia. Histologically, the cyst wall was lined by a columnar pseudo-stratified and squamous epithelium. Good cosmetic results without recurrence were achieved during the 6-month postoperative period. A parameatal cyst should be treated with complete surgical excision. Puncture of the cyst with a needle or surgical incision may result in recurrence.
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