Median raphe cyst of the perineum is a rare congenital anomaly and was first reported in 1895. We report a 9-year-old male with median raphe cysts in the perineum which had become swollen and then spontaneously ruptured several times since he was 3 years old. There were two separate lesions; one was a cystic lesion and the other was a canal-like lesion. They were completely excised and the inner layer of the cysts was composed of transitional epithelium.
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