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We report a case of primary extragonadal germ cell tumor of the penis in a 2-year-old child with elevated serum alpha-fetoprotein. The patient responded well to a combination of bleomycin, etoposide and cisplatin, achieving a complete response within 3 cycles. He was alive and disease-free 8 months after diagnosis. To our knowledge this is the first case of an endodermal sinus tumor of the penis reported in the literature. The possible histogenesis of this tumor at such a rare site is discussed.
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