Surface epithelial tumors account for more than 90% of ovarian tumors, of which serous tumors comprise 46%. They are usually cystic but sometimes may present with solid, fibrous component and papillary excrescences which are then called papillary serous cysadenofibroma.

A multiparous patient of age 32 years reported with lower abdominal distension since 1 month and pain abdomen since a day. On per abdominal examination there was a mass corresponding to 16weeks gravid uterus with irregular surface and free side to side mobility. The lower border could not be reached, thus seem to arise from pelvis. There was tenderness in right iliac fossa with guarding. Per vaginal examination showed bilateral forniceal fullness with tenderness in right fornix. Ultrasonography showed a bilateral complex cystic lesion arising from ovaries with torsion on right side. Emergency Laparotomy was done and on frozen section the tumor was confirmed to be of benign nature. Right side salphingo-oophrectomy was done along with left side ovarian cystectomy was done. Histopathological examination of the specimen confirmed it to be bilateral serous papillary cystadenofibroma with torsion on right side.

Serous papillary cystadenofibroma usually presents as a complex ovarian cyst with solid and cystic components and irregular septations, due to which is often misdiagnosed as malignant tumor per-operatively. Management is mainly surgical removal of cyst with or without ooptorectomy. Frozen section aid in the confirmation of its benign nature and to avoid unnecessary extensive surgery.

Keywords: Serous papillary cystadenofibroma, Ovarian complex cyst, Torsion of ovarian cyst, Frozen section, Cystectomy 

Serous tumors of the ovaries accounts to one fourth of all ovarian tumors. It occurs in the age group of 15-65 years. Serous cysadenofibroma is one such tumor, which although is benign in nature masquerade as malignant on imaging, perioperatively and grossly [1]. It is a rare benign ovarian tumor containing both epithelial and fibrous stromal component. It is a slow growing tumor but appears as complex mass lesion with both solid and cystic component on ultrasonography, thus easily confused to that of malignant ovarian pathology [2]. Histopathological examination of the specimen helps in differentiating it from that which is truly malignant.

The main presenting symptom of Serouscysadenofibroma is mass per abdomen, bleeding per vagina and pain abdomen. Here we present a rare case of bilateral papillary serous cystadenofibroma in a 32 years old patient presenting with mass per abdomen followed with pain abdomen. This case signifies the importance of microscopy in neoplasms that masquerade as malignant on imaging, perioperatively and grossly, thus achieving excellent prognosis with surgery alone.

CASE REPORT

A multiparous patient of age 32 years reported to the gynecology OPD in ESIC MC PGIMSR, Rajajinagar, Bangalore with lower abdominal distension since 1 month and pain abdomen since one day. There was no history of significant weight loss or loss of appetite. The patient did not have any disturbances in bowel or bladder habits or associated medical co morbidities. The patient had regular menstrual cycles.

On examination the patient was moderately built and nourished with a BMI of 24.3 kg/m². The patient had tachycardia with pulse rate of 98 beats/min. Other vitals were within normal limits. On per abdominal examination there was a mass corresponding to 16weeks gravid uterus with irregular surface and free side to side mobility. The lower border could not be reached, thus seem to arise from pelvis. There was tenderness in right iliac fossa with guarding. Per vaginal examination showed bilateral forniceal fullness with tenderness in right fornix.

Ultrasonography showed a bilateral complex cystic lesion arising from ovaries, right sided complex cyst measuring 20 × 14 cm with torsion and a left sided complex cyst measuring 11 × 6 cm. Haematological investigations were within normal limits. Serum CA-125 was 66.9 IU/L (Figure 1).

The patient was posted for emergency Laparotomy proceeded by Right side salphingo-oophrectomy was done along with left side ovarian cystectomy was done (Figure 2). Frozen section was performed on the specimen and found to be of benign nature. Histopathological examination of the specimen confirmed it to be bilateral serous papillary cystadenofibroma with torsion on right side. The post-operative period was uneventful and patient was discharged on POD-7. Follow up of patient for one year did not show any recurrence.

Patient was advised to take calcium with d3 supplementation, regular walking, exercise and repeat CA-125 was 15.9 IU/L. In view of benign nature of the serous tumor the patient was followed with CA-125. Although mucinous tumors of ovary has higher recurrence rate.

DISCUSSION

Surface epithelial tumors account for more than 90% of ovarian tumors, of which serous tumors comprise 46%. They are usually cystic but sometimes may present with solid, fibrous component and papillary excrescences which are then called papillary serous cysadenofibroma [3]. Other variants of serous epithelial tumors depending on the amount of fibrous tissue present in it can be classified into cystadenoma, papillary cystadenoma, papillary cystadenofibroma and papillary adenofibroma. Microscopically they are lined by a single layer of ciliated columnar epithelium or cuboidal non-ciliated epithelium. Papillary projections with stroma having varying degree of fibroblasts are seen [4]. Because of their solid component or irregular septations these tumors mimic malignancy on gross appearance and on ultrasonography.

In a study by Cho et al. [5], 16 cases of ovarian cystadenofibroma which presented with complex cystic masses were preoperatively misdiagnosed as malignant tumors on CT scan or MRI. Thus a perioperative frozen section is helpful in correct diagnosis and reduces the need of unnecessary extensive surgery.

It mainly presents with abdominal pain, lower abdominal distension, dysuria, bowel disturbances, vaginal bleeding and feminization [6]. Vaginal bleeding and feminization are due to excessive estrogen secretion by the tumor causing abnormal endometrial growth [7].

Management is mainly surgical with favorable outcome with cystectomy alone or alone with salpingo-oophorectomy [8]. Sills et al. [9], in their study described the technique of decompression and intact removal of cyst without incidence through a 5 mm laparoscopic cannuala.

CONCLUSION

Papillary serous cystadenofibroma of ovary is a rare benign tumor seen in the age group of 15-65 years. It usually presents as a complex ovarian cyst with solid and cystic components and irregular septations, due to which is often misdiagnosed as malignant tumor per-operatively. Management is mainly surgical removal of cyst with or without ooptorectomy. Frozen section aid in the confirmation of its benign nature and to avoid unnecessary extensive surgery. The overall prognosis with this tumor is excellent. 

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7.       Bell DA, Scully RE (1985) Benign and borderline clear celladenofibromas of the ovary. Cancer 56: 2922-2931.

8.       Czernobilsky B, Borenstein R, Lancet M (1974) Cystadenofibroma of the ovary. Cancer 34: 1971-1981.

9.       Sills ES, Kaplan CR, Perloe M, Tucker MJ (2003) Laparoscopic approach to an uncommon adnexal neoplasm associated with infertility: Serous cystadenofibroma of the fallopian tube. J Am Assoc Gynecol Laparosc 10: 545-547.