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 Table of Contents 
CASE REPORT
Year : 2014  |  Volume : 5  |  Issue : 3  |  Page : 156-158  

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


1 Department of Obstetrics and Gynecology, University College of Medical Sciences and Guru Teg Bahadur Hospital, New Delhi, India
2 Department of Pathology, University College of Medical Sciences and Guru Teg Bahadur Hospital, New Delhi, India

Date of Web Publication19-Sep-2014

Correspondence Address:
Dr. Rajlaxmi Mundhra
Department of Obstetrics and Gynecology, Guru Teg Bahadur Hospital, New Delhi - 110 095
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0976-7800.141227

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   Abstract 

Xanthogranulomatous inflammation is a rare, non-neoplastic variant of chronic inflammation. Of the 15 reported cases of Xanthogranulomatous endometritis, majority (60%) were accompanied by endometrial carcinoma. We herein report a case of a postmenopausal woman presenting with recurrent pyometra who was suspected to be case of cervical tuberculosis and treated for the same. Inspite of antitubercular therapy, she had cervical stenosis and developed recurrent pyometra over the next 2 years requiring repeated drainage. She then underwent hysterectomy and was found to have co-existent Xanthogranulomatous salpingitis (XGS) on histopathological examination. Xanthogranulomatous inflammation is a rare pathological diagnosis that gynecologists need to be aware of while managing such patients.

Keywords: Recurrent pyometra, salpingitis, xanthogranulomatous inflammation


How to cite this article:
Gami N, Mundhra R, Guleria K, Arora VK, Garg S. Recurrent pyometra and xanthogranulomatous salpingitis: A rare pathologic association in a postmenopausal lady. J Mid-life Health 2014;5:156-8

How to cite this URL:
Gami N, Mundhra R, Guleria K, Arora VK, Garg S. Recurrent pyometra and xanthogranulomatous salpingitis: A rare pathologic association in a postmenopausal lady. J Mid-life Health [serial online] 2014 [cited 2021 Dec 3];5:156-8. Available from: https://www.jmidlifehealth.org/text.asp?2014/5/3/156/141227


   Introduction Top


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.


   Case Report Top


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.
Figure 1: Pap smear showing histiocytic granuloma

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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.
Figure 2: Hysterectomy specimen showing granuloma in right tube

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Pathology

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.
Figure 3: 40× photomicrograph of cholestrol clefts surrounded by xanthomous cells and chronic inflammation

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The post-operative period was unremarkable and the woman was discharged on 6 th post-operative day with an advice to follow-up.


   Discussion Top


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.

 
   References Top

1.Kunakemakorn P, Ontai H, Balin H. Pelvic inflammatory pseudotumor: A case report. Am J Obstet Gynecol 1976;26:286-7.  Back to cited text no. 1
    
2.Noack F, Briese J, Stellmacher F, Hornung D, Horny HP. Lethal outcome in xanthogranulomatous endometritis. APMIS 2006;114:386-8.  Back to cited text no. 2
    
3.Franco V, Florena AM, Guarneri G, Gargano G. Xanthogranulomatous salpingitis. Case report and review of the literature. Acta Eur Fertil 1990;21:197-9.  Back to cited text no. 3
    
4.Mittal BV, Badhe BP. Xanthogranulomatous pyelonephritis - (a clinicopathological study of 15 cases). J Postgrad Med 1989;35:209-14.  Back to cited text no. 4
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5.Zhang XS, Dong HY, Zhang LL, Desouki MM, Zhao C. Xanthogranulomatous inflammation of the female genital tract: Report of three cases. J Cancer 2012;3:100-6.  Back to cited text no. 5
    
6.Cozzutto C. Xanthogranulomatous osteomyelitis. Arch Pathol Lab Med 1984;108:973-6.  Back to cited text no. 6
[PUBMED]    
7.Usamentiaga E, Val-Bernal JF, Alonso- Bartolome´ P, Lo´pez-Rasines G, del Valle JI, Calabia A. Xanthogranulomatous orchitis. Urology 1998;52:891-2.  Back to cited text no. 7
    
8.Gray Y, Libbey NP. Xanthogranulomatous salpingitis and oophoritis: A case report and review of the literature. Arch Pathol Lab Med 2001;125:260-3.  Back to cited text no. 8
    
9.Howey JM, Mahe E, Radhi J. Xanthogranulomatous salpingitis associated with a large uterine leiomyoma. Case Rep Med 2010;2010:970805.  Back to cited text no. 9
    
10.Santamaría IB, Ulacia MA, Perez JM. Endometritis xantogranulomatosa. Rev Esp Patol 2004;37:419-22.  Back to cited text no. 10
    
11.Shashikala K, Sharmila PS, Sushma TA, Francis P. Ovarian haemangioma with synchronous xanthogranulomatous inflammation-a rare pathological finding. Int J Health Sci Res 2013;3:116-9.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]


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