Imperforate hymen is a urogenital tract anomaly that represents the most frequent congenital malformation of the female genital tract. CA 19-9 and CA 125 are widely used as tumor markers, however several benign conditions are also known to increase serum CA 19-9 and CA 125 levels. According to classical textbook knowledge, imperforate hymen is not listed under the benign conditions that increase serum CA 19-9 and CA 125 levels. In this case report we describe a relation between imperforate hymen and elevated serum CA 19-9 and CA 125 levels.