Xanthogranulomatous inflammation in female pelvis: a diagnostic and therapeutic challenge

Abstract

Xanthogranulomatous inflammation is an unusual form of chronic inflammation that can present clinically with destructive or obstructive symptoms. It may also mimic malignancy at times. It is a rare entity as far as female genital tract is concerned. Histologically well characterized by a massive infiltration of the tissues by lipid-laden histiocytes, it usually poses diagnostic and therapeutic challenges for the clinician. We present here two interesting cases of the same, encountered by us. Case 1: A 42-year-old lady with history of intra-uterine device (IUD) in situ for 8 years presented with a picture of acute on chronic pelvic inflammatory disease (PID). She was started on inpatient CDC regimen of PID. Despite this her symptoms persisted and based on high index of suspicion she was started empirically on anti-tubercular treatment (ATT). Three months later she presented again with similar complaints. Laparotomy picture was typical of abdominal tuberculosis and thus multiple biopsies were obtained. Histo-pathological report showed xanthogranulomatous inflammation. As there is no specific treatment for this condition, patient was continued with ATT. On three, six and twelve months follow up patient had been asymptomatic. Case 2: A 48 year old lady with history of vaginal hysterectomy 6 months ago presented with right sided hydro-uretero nephrosis. CT scan showed this was because of external compression caused by an adnexal mass measuring 5.8×3.9 cms. She underwent laparotomy with right oophorectomy. Right distal ureter was fibrotic so was excised and Boari flap was done. Histopathological diagnosis rendered was Xanthogranulomatous inflammation with abscess formation and chronic ureteritis with fibrosis. Xanthogranulomatous inflammation of the genital tract is enigmatic disorder of unknown etiology. Gynecologist should be aware of this entity whenever the clinical presentation is atypical and not fitting into a diagnosis, or when the patient is not responding to conventional treatment of PID. Early diagnosis and management is very important as it can cause tissue destruction or may lead to obstructive disasters.