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To the best of our knowledge, there are currently no reports on primary uterine ependymoma. We operated on a 38‐year‐old woman suspected with uterine fibroids; surgical findings led to the diagnosis of primary anaplastic ependymoma of the uterus. Most extraneural ependymomas arising from the ovary and perigenital peritoneum occur in women of reproductive age and express estrogen and progesterone receptors...