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  Table of Contents    
CASE REPORT  
Year : 2011  |  Volume : 54  |  Issue : 1  |  Page : 141-143
Xanthogranulomatous salpingitis as a rare pathologic aspect of chronic active pelvic inflammatory disease


1 Department of Pathology, Maltepe University, Istanbul, Turkey
2 Department of Obstetrics and Gynecology, Maltepe University, Istanbul, Turkey

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Date of Web Publication7-Mar-2011
 

   Abstract 

Xanthogranulomatous salpingitis (XGS) is a rare form of chronic inflammation of the fallopian tubes. A 41-year old woman with a history of secondary infertility for 2 years is presented. The patient underwent bilateral salpingooopherectomy with presumptive diagnosis of adnexal mass with cystic component. Intraoperative pathology consultation was done. The diagnosis of bilateral XGS associated with chronic active follicular salpingitis was made. XGS is reported to be caused by an unsuccesfully treated pelvic inflammatory disease. Its association with chronic active follicular salpingitis has not been previously reported. Chronic active follicular salpingitis with xanthogranulomatous inflammation might give the impression of a cystic adnexal mass with septations on preoperative pelvic computed tomography. Frozen sections are necessary to rule out malignancy as done in our case.

Keywords: pelvic inflammatory disease, salpingitis follicularis, xantho-granulomatous salpingitis

How to cite this article:
Yener N, Ilter E, Midi A. Xanthogranulomatous salpingitis as a rare pathologic aspect of chronic active pelvic inflammatory disease. Indian J Pathol Microbiol 2011;54:141-3

How to cite this URL:
Yener N, Ilter E, Midi A. Xanthogranulomatous salpingitis as a rare pathologic aspect of chronic active pelvic inflammatory disease. Indian J Pathol Microbiol [serial online] 2011 [cited 2019 Nov 21];54:141-3. Available from: http://www.ijpmonline.org/text.asp?2011/54/1/141/77373



   Introduction Top


Xanthogranulomatous salpingitis (XGS) is a rare, destructive type of inflammation of unknown cause characterized by an accumulation of excessive foamy macrophages with accompanying lymphocytes, plasmocytes, and a few polymorphonuclear leukocytes. [1] Rare xanthogranulomatous inflammation of the  Fallopian tube More Detailss have also been reported. [2],[3],[4],[5],[6] As elsewhere in the body, the mechanism of this form of inflammatory response in the fallopian tubes is not clear.

In this study, we report a case of XGS with overt tubal follicle-like structure formation that gave the impression of an adnexal mass with cystic component. This rare form of inflammation of the fallopian tube is discussed with differential diagnoses and a review of the relevant literature.


   Case Report Top


A 41-year-old woman (gravida 1, parity 1) was admitted to our emergency clinic with a complaint of bilateral groin pain for 6 months that gradually increased over the last 2 weeks. The patient was not in a toxic state. Although she and her husband were not using any contraceptive method, she was infertile for nearly 2 years. But they did not receive professional help.

In the gynecologic investigation, there was bilateral adnexal tenderness and the transvaginal sonography showed the uterus as normal and a cystic mass with a size of 39 × 48 mm at the right adnexal region and another one with a size of 33 × 22 mm on the left. A pelvic computedtomography was performed, suggesting bilateral adnexal lesions with multiple cystic component with the biggest size of 40 × 35 mm at the right, 35 × 30 mm at the left [Figure 1]. It was also reported that both of the cystic masses contained septations. Radiologically, the adnexal masses on both sides were thought to represent either a tubal or an ovarian neoplasm with cystic component. The laboratory findings including the tumor markers were all in normal ranges. The patient's serum cholesterol was within normal limits as well. The patient underwent exploratory laparotomy with the presumptive diagnosis of bilateral adnexal cystic neoplasm. On exploration, the uterus and both ovaries were normal but there were bilateral hydrosalpinges. Bilateral salpingectomy was performed and both ovaries were wedge biopsied. Intraoperative pathology consultation was reported as benign. After achieving hemostasis, the operation was completed without any early complication. The patient made an uneventful recovery and was discharged home on the second postoperative day. She did not undergo any adjuvant therapy and has been doing well for the last 2 months after her operation.
Figure 1: Pelvic computed tomography showing salpinges that mimic bilateral adnexal masses with cystic components

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   Pathology Top


On gross examination, the fallopian tubes were bilaterally enlarged [Figure 2]a and b. The left tube with dimensions of 7 × 1.7 × 1.5 cm exhibited dilated lumina with inspissated mucus and a yellow-orange material inside [Figure 2]a. The left ovary contained corpus luteum. The right tube measured 6 × 1.5 × 1.5 cm and contained a yellow-orange material that completely filled the lumen [Figure 2]b. The right ovary contained a typical follicle cyst. Both fallopian tubes were entirely grossed in. Bilateral tubes and the ovaries were entirely sectioned, submitted, and examined. Microscopically, both tuba uterinas revealed similar morphologic findings. Tubal plicae on both sides were fused and revealed follicle-like dilated structures lined by tubal epithelium in many areas. The tubal lumina were completely filled with both acute and chronic inflammatory cells composed of mainly xanthomatous cells admixed with plasma cells, lymphocytes, and few neutrophils [Figure 3]a and b. Floret-like multinucleated foamy macrophages were also present [Figure 3]c and d. Large areas of tubal mucosa were destroyed and the tubal epithelium was replaced by organized luminal protrusions composed of the inflammatory reaction [Figure 4]. Almost all sections of both tubes revealed follicle-like dilated structures lined with tubal epithelium, and the xanthogranulomatous inflammation was also noticed around these structures [Figure 3]a. These follicle-like structures extended to the muscular or even serosal layer of both tubes. There was no sign of granulomata, foreign bodies, or parasitic or fungal infections on both sides. The diagnosis of xanthogranulomatous inflammation with active chronic follicular salpingitis was made.
Figure 2: Enlarged left (a) and right (b) uterine tubes on gross examination

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Figure 3: (a and b) Xanthogranulomatous reaction filling the right tubal lumina and existing throughout the wall associated with follicular salpingitis (H and E, ×40); (c-d) foamy macrophages with some floret-like multinucleated forms predominate (H and E, ×400). (*) indicates tubal lumina in (a)

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Figure 4: Tubal mucosa destruction and organized luminal protrusions composed of xanthogranulomatous reaction in the left uterine tube (H and E, ×40)

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   Discussion Top


Xanthogranulomatous inflammation has been described in several organs, including those of the female genital tract. Pelvic endometriosis, [3],[6] pelvic inflammatory disease (PID), [3],[4],[5],[6] intrauterine device (IUD) and chronic endometritis, [2] and chemotherapy due to a breast malignancy [2] have all been suggested as causes for this uncommon form of inflammatory response in the fallopian tubes. To this date, only a small number of cases of xanthogranulomatous inflammation occurring in the fallopian tubes have been described. [2],[3],[4],[5],[6] In previously reported cases, the morphology was similar. In each case the tubal plicae were distended and thickened with the xanthogranulomatous inflammation. Interestingly, we observed a different histopathologic finding in our case. These included blunted and fused mucosal plicae and extensive follicle-like structure formations in many areas of the tubal mucosa. This is a well-known entity called "salpingitis follicularis." It is seen in the process of chronic active salpingitis. [7] These follicle-like structures are the result of the adherence of mucosal plicae to one another because of the continuous surface fibrin deposition that leads to permanent bridging between folds. In our case, we observed this phenomenon in association with XGS demonstrating the latter as a pathologic aspect of chronic active PID.

A possible explanation for this coexistence is that the chronic infection leads to tissue necrosis. This in turn, continuously releases cholesterol and other lipids from the dead cells. Then these cell constituents are phagocytosed by macrophages, leading to xanthomatous process.

Radiologically, salpingitis follicularis (SF) may mimic an adnexal mass with cystic component, as in our case. Histopathologic examination of the material should be done to make a final diagnosis. Another differential diagnosis of these follicle-like structures is salpingitis isthmica nodosa (SIN), which is a diverticular disease of the fallopian tubes that causes infertility. [8],[9] The etiology of SIN is unknown; the possible explanations are postinflammatory distortion and adenomyosis-like process. However, in contrast to the SF, SIN typically appears as yellow-white nodular swellings located at the isthmic portion of the tube. Microscopically they represent the diverticula-like structures that lie within the hyperplastic smooth muscle with scant surrounding inflammation. [8],[9]

While endometriosis is also suggested to cause XGS, [2],[3] cystic endometrial glands of tubal endometriosis should also be considered in the differential diagnosis. However, endometrial glands are lined by columnar epithelium without having any cilias typical for tubal epithelium. In our case we submitted both left and right tubes entirely and did not notice any focus of endometriosis.

In conclusion, we emphasized the possible etiologic relationship between XGS and chronic active follicular salpingitis. It should be borne in mind that XGS may also cause follicle-like structures and may be preoperatively misdiagnosed as an adnexal neoplasm hence frozen section is mandatory to rule out malignancy.


   Acknowledgment Top


We would like to thank Mr Michael Robinson from Colorado for his language assistance.

 
   References Top

1.Adsay VN. Gallbladder, extrahepatic biliary tree, and ampulla. In: Mills SE, editor. Sternberg's Diagnostic Surgical Pathology. Philadelphia: Lippincott Williams and Wilkins; 2004. p.1786.  Back to cited text no. 1
    
2.Idrees M, Zakashansky K, Tamara K. Xanthogranulomatous salpingitis associated with fallopian tube mucosal endometriosis: A clue to the histogenesis. Ann Diagn Pathol 2007;11:117-21.  Back to cited text no. 2
    
3.Gray Y, Libbey P. 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. 3
    
4.Furuya M, Murakami T, Sato O, Kikuchi K, Tanaka S, Shimizu M, et al. Pseudoxanthomatous and xanthogranulomatous salpingitis of the fallopian tube: A report of four cases and a literature review. Int J Gynecol Pathol 2001;21:56-9.  Back to cited text no. 4
    
5.Punia RS, Aggarwal R, Amanjit MH. Xanthogranulomatous oophoritis and salpingitis: Late sequelae of inadequately treated staphylococcal PID. Indian J Pathol Microbiol 2003;46:80-1.   Back to cited text no. 5
    
6.Singh N, Dadhwal V, Sharma KA, Mittal S. Xanthogranulomatous inflammation: A rare cause of premature ovarian failure. Arch Gynecol Obstet 2009;279:729-31.  Back to cited text no. 6
    
7.Wheler JE. Diseases of the Fallopian tubes. In: Kurman RJ, editor. Blaustein's Pathology of the female genital tract. 5 th ed. Springer: New York, 2002; p. 624.  Back to cited text no. 7
    
8.Chawla N, Kudesia S, Azad S, Singhal M, Rai SM. Salpingitis isthmica nodosa. Indian J Pathol Microbiol 2009;52:434-5.  Back to cited text no. 8
[PUBMED]  Medknow Journal  
9.Creasy JL, Clark RL, Cuttino JT, Groff TR. Salpingitis isthmica nodosa: Radiologic and clinical aspects. Radiology 1985;154:597-600.  Back to cited text no. 9
    

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Correspondence Address:
Nese Yener
Department of Pathology, Maltepe University Hospital, 34843, Maltepe, Istanbul
Turkey
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.77373

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]

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    Abstract
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