Background: Benign serous papillary cystadenofibroma (SPCAF) of the ovary is a very rare subtype of serous tumors of the ovary. Commonly occurs between 15 and 65 years of age. They are usually asymptomatic or diagnosed as an incidental finding. Routine imaging features masquerade malignancy and lead to extensive surgeries. Intraoperative frozen section helps in avoiding radical surgeries. Case description: A 28-year-old multiparous, sterilized woman presented with complaints of lower abdominal pain, but no menstrual complaints. Abdominal examination revealed 14 × 14 cm solid with cystic lesion occupying lower abdomen. Ultrasound (US) and contrast enhanced computed tomography (CECT) of the abdomen showed a complex left ovarian cyst with torsion. CA 125 was extremely elevated 3646 U/mL. Other tumor markers were normal. A staging laparotomy was done. Intra-operative findings showed the left ovary replaced by 14 × 14 cm cyst, which underwent torsion 2 times. The ovarian cyst was untwisted and a left salpingo-oophorectomy was done. The cut section of the specimen showed papillary projections of 2 × 2 cm over the inner wall of the cyst. The frozen section histopathology report was benign SPCAF. Hence radical surgery was avoided. The final histopathology report revealed benign SPCAF of the left ovary. Conclusion: This case is presented in view of its rarity and atypical presentation. The presentation was atypical, since CA 125 was extremely elevated and imaging features suggested a malignant neoplasm. But intraoperative frozen section histopathology turned out to be a benign tumor thus avoiding radical surgery in a young patient. Clinical significance: Diagnosis of benign SPCAF ovary is difficult as imaging features masquerade malignancy, hence intraoperative frozen section plays a crucial role in definitive diagnosis and helps in avoiding extensive surgeries.