Recurrent pyometra and xanthogranulomatous salpingitis: A rare pathologic association in a postmenopausal lady.

60-years-old, P 7 L 7 , postmenopausal lady presented with complaints of foul smelling pus like discharge per vaginum for 3 months. She was a known hypertensive and is on regular medication. General physical and abdominal examination was unremarkable. Per Speculum examination revealed an inflamed cervix, flushed with the vagina and pus like discharge coming through os. On per vaginal examination, uterus was bulky, bilateral fornices were free. Pelvic ultrasound revealed intrauterine collection. Pap smear was taken and cervical dilatation with pyometra drainage was done. Around 5 cc pus was drained. Pus culture was sterile and negative for acid fast bacilli. Pap smear showed granulomatous cervicitis [ Figure 1 ] and based on it the patient was started on anti-tubercular therapy for 6 months. Nine months later, the woman presented with blood mixed discharge per vaginum and underwent dilatation and curettage which suggested chronic endometritis. A repeat Pap smear was done and it showed inflammatory smear. Antibiotic course was given with an advice to follow-up. Pap smear showing histiocytic granuloma A year later, she presented again with pyometra. Drainage was done. Culture was sterile and negative for acid fast bacilli and she continued to have pus discharge. Ultrasound was repeated which showed 100 cc intrauterine collection. Repeat drainage was done, for the third time, and an intrauterine Foleys catheter no 8 was put for continuous drainage. Pus culture showed E. Coli infection and based on sensitivity, antibiotics were started. In view of recurrent pyometra, a differential of tubercular endometritis or endometrial malignancy was suspected. In view of recurrent pyometra requiring repeated drainage she was taken up for hysterectomy with bilateral salpingo-oopherectomy. The abdominal route was planned as the uterus was very soft and cervix was flushed with the vagina. During the surgery, it was seen that the uterus was very soft and bulky. Bilateral tubes were studded with tubercles. A 2 × 1 cm solid, yellow nodule was present near the corneal end of right tube, which on cut section showed exudation of caseous purulent material [ Figure 2 ]. Clinical suspicion of fallopian tube malignancy was made. Hysterectomy specimen showing granuloma in right tube The gross examination of the specimen revealed dilated right fallopian tube measuring 5.5 cm in length with a solid 2 × 1 × 0.5 cm yellow colored nodule at the cornual end. The left fallopian tube measured 4.5 cm in length with unremarkable uterus and cervix. The bilateral ovaries showed corpus luteum. The histopathological examination of bilateral fallopian tubes showed acute and Xanthogranulomatous inflammation, composed of mainly foamy histiocytes, lymphocytes, plasma cells and few neutrophils [ Figure 3 ]. Sections from uterus showed surface ulceration with chronic endometritis. The bilateral ovaries were unremarkable. 40× photomicrograph of cholestrol clefts surrounded by xanthomous cells and chronic inflammation The post-operative period was unremarkable and the woman was discharged on 6 th post-operative day with an advice to follow-up.

Xanthogranulomatous inflammation is an uncommon form of chronic inflammation, causing destruction of affected organs and is characterized by the presence of lipid laden histiocytes admixed with lymphocytes, plasma cells and leucocytes.[ 3 ] It usually involves kidneys and gall bladder.[ 4 ] Involvement of female genital tract as such is rare but when it does so, it is the endometrium which usually gets affected. To our knowledge, no case of Xanthogranulomatous salpingitis (XGS) has been reported in association with recurrent pyometra.

Xanthogranulomatous inflammation is a rare, chronic inflammation affecting the organs of the female genital system. XGS mainly involves unilateral fallopian tubes, occasionally occurs bilaterally. It is clinically often accompanied with inflammation in vagina, cervix and endometrium. The visibly thickened fallopian tube is variable in length, with an appearance of mass lesion. There is exudation on the serosal surface and the fimbriated end of fallopian tube is usually obliterated. Morphologically, the fallopian tube structure remains intact.[ 5 ] The exact etiology has remained elusive but it has been associated with infection, ineffective antibiotic therapy leading to ineffective clearance of bacteria by phagocytes, endometriosis and pelvic inflammatory disease.[ 6 7 ] Organisms such as E coli, Proteus spp., Staphylococcus aureus, Bacteriodes fragilis and Salmonella typhi have been isolated from bacterial culture.[ 8 ] In our case of recurrent pyometra, a combination of factors may have been responsible for the development of Xanthogranulomatous inflammation of the fallopian tubes. Granulomatous cervicitis, probably secondary to tuberculosis led to cervical stenosis. This led to recurrent pyometra in the lady. Chronic collection of pus in the uterine cavity leading to obstruction of the fallopian tubes may have predisposed to infection in the tubes followed by tissue necrosis and release of cholesterol and other lipids, and phagocytosis by macrophages. Also we had found infection with E coli which has been found to be associated with Xanthogranulomatous inflammation. A review of the literature done by Margaret et al ., revealed that patients with XGS ranged from 23-72 years old and initial presentation most often included lower abdominal or suprapubic pain, fever, menorrhagia or vaginal bleeding, and physical examination typically showed a pelvic mass with adnexal tenderness.[ 9 ] But this is probably the first reported case of XGS found in association with recurrent pyometra. Pyometra was seen to be associated with Xanthogranulomatous endometritis in 74-year-old woman in a case reported by Santamaria et al .[ 10 ] Pyometra was found in association with Xanthogranulomatous endometritis was also seen in a 69-year-old woman with a short history of abdominal pain and a palpable mass in the pouch of Douglas.[ 2 ] A case of cavernous ovarian hemangioma with xanthogranulomatous salpingooophoritis in the contralateral adnexa has also been reported in a 61-year-old patient presenting with postmenopausal bleeding and pain abdomen.[ 11 ] The importance of Xanthogranulomatous inflammation lies in the fact that it can present as a lump or tumor and can even mimic malignancy. Hence, gynecologists need to be aware of this entity and be able to differentiate it from endometrial, ovarian or tubal malignancy. Untreated Xanthogranulomatous inflammation has also been reported to be lethal by causing systemic inflammation.[ 2 ] Hence, aggressive treatment in the form of surgery should be strongly considered, if diagnosed pre-operatively. In our case, timely hysterectomy prevented any progress of the condition and prevented systemic inflammation and its consequences. Xanthogranulomatous inflammation is a rare condition affecting the female genital system. It should be borne in mind while managing chronic infective conditions like recurrent pyometra, as it has been known to progress to systemic inflammation which can be lethal.