Xanthogranulomatous salpingo-oophoritis presenting as an ovarian tumour

Background

Xanthogranulomatous inflammation results from the accumulation of lipid-laden foamy macrophages with lymphocytes, neutrophils and plasma cells along with multinucleated giant cells.1 It is responsible for chronic inflammation, which leads to the destruction of the affected organ, with kidney being the most commonly affected organ.2 Xanthogranulomatous inflammation affecting the female genital tract is extremely rare and usually involves the endometrium, fallopian tube and ovaries.3 Reported cases usually present as a pelvic inflammatory disease that does not respond to antibiotics or as a mass lesion simulating adnexal malignancy.4 Here we report a case of xanthogranulomatous salpingo-oophoritis (XGSO) in a woman who was operated with suspicion of a borderline ovarian tumour.

Case presentation

A nulliparous woman attended at gynaecological service with intermittent lower abdominal pain of 3 months duration without other complaints. The pain was more intense in the left iliac fossa, it did not radiate and worsened with sexual intercourse. She refused any vaginal discharge or abnormal bleeding, fever, nausea, vomiting, urinary problems or bowel habit changes. She had menarche at 13 years old, with regular menses. There was no preceding pathological history of importance. General examination revealed good general condition, with normal vital sings, coloured and hydrated mucous membranes. Abdominal examination revealed a soft and non-tender abdomen and there was no palpable mass. On per vaginal examination, a firm left adnexal mass could be palpated.

Investigations

Ultrasonography (USG) showed a left adnexal mass measuring 88×75 mm, with low flux resistance vascularisation and multiple septations. Her complete blood count was within a normal range. Cancer antigen 125 (CA-125) was 35.2 U/mL, carcinoembryonic antigen was 1.05 mg/mL, Ca 19.9 was 21 mg/mL and beta-human chorionic gonadotropin <2 U/mL. The patient was submitted to a left adnexectomy with frozen section histopathology in view of suspected borderline ovarian tumour.

Differential diagnosis

Differential diagnosis of an adnexal mass with 8 cm and multiple septations includes an ovarian tumour, endometrioma, tubo-ovarian abscess, tuberculosis and fungal infections.

Neoplastic lesions can be ruled only in histopathological examination. Abscess, tuberculosis and fungal infections can be confirmed by culture and special stains for the causative organisms.

Treatment

An exploratory laparoscopy with extensive adhesyolisis was performed. There was a mass measuring 8 cm on the left adnexa, which was densely adhered to the omentum and large bowel. The patient underwent a left salpingo-oophorectomy. The frozen section examination revealed tissue extensively infiltrated by histiocytoids, plasma cells and lymphocytes with necrosis—an aspect suggestive of a chronic and xanthomatous inflammatory process. No unmistakable evidence of malignancy. Final histopathological examination (figure 1A–D) showed ovarian and fallopian tube with dense granulation tissue formation with abundant foamy macrophages, surrounded by fibrocollagenous tissue showing infiltration by lymphoplasmacytic cells and macrophages, along with congested blood vessels. Thus, a final diagnosis of XGSO was given.

Outcome and follow-up

The patient had an uneventful postoperative recovery and was discharged on the third postoperative day. Ten days later, the patient returned to the gynaecological service with fever, abdominal pain and diarrhoea. Her abdomen was distended, slightly depressing and painful in the lower quadrants. Transvaginal ultrasound revealed a palpable pelvic swelling between the pouch of Douglas and the umbilical area. Pelvic USG revealed a pelvic swelling surrounding the uterus. She was hospitalised again and underwent antibiotic therapy with clindamycin and gentamicin. Pelvic abscess was submitted to drainage by intervention radiology with favourable outcome.