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Sertoli-Leydig cell tumor (SLCT) of the ovary is an extremely uncommon neoplasm accounting for &lt;0.5% of all primary ovarian neoplasms. These tumors belong to the category of sex cord-stromal tumors. The tumor has variable clinical and histopathological presentations complicating the diagnosis and therefore the treatment. The presence of heterologous elements is seen in one-fifth of these already rare neoplasms. Herein, we report a case of a 28-year-old female presenting with irregular menses, features of virilization, and abdominal pain. Histopathological examination revealed marked focal anaplasia in this tumor of, otherwise, intermediate differentiation along with the presence of heterologous elements. Reporting of such elements is imperative for adequate treatment and deciding follow-up.

Sertoli-leydig cell tumor of ovary: A rare case report with heterologous elements and focal marked anaplasia