Xanthogranulomatous oophoritis: A case report with review of literature

Suparna Milind Bindu; Meera S Mahajan

doi:10.4103/2278-344X.138604

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CASE REPORT

Year : 2014 | Volume : 3 | Issue : 3 | Page : 187-189

Xanthogranulomatous oophoritis: A case report with review of literature

Suparna Milind Bindu, Meera S Mahajan
Department of Pathology, MGM Medical College, Aurangabad, Maharashtra, India

Date of Web Publication

13-Aug-2014

Correspondence Address:
Suparna Milind Bindu
Department of Pathology, MGM Medical College, Aurangabad, Maharashtra
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/2278-344X.138604

Abstract

Xanthogranulomatous inflammation is an uncommon form of chronic inflammation in which the affected organ is destroyed and is replaced by large number of lipid - containing macrophages with an admixture of lymphocytes, plasma cells and multinucleated giant cells. The most commonly affected organs are the kidney and gall bladder, followed by anorectal area, bone, stomach, and testis. The inflammation most often affects the endometrium in the female genital tract, but involvement of the vagina, cervix, fallopian tube, and ovary may also occur. Only a few cases involving the ovary have been reported. We report a case of xanthogranulomatous infection of ovary in a 40-year-old female who presented with abdominal pain and bleeding per vaginum. Her clinical examination, radiological evaluation, surgical findings and gross features were likely to be mistaken for malignancy if xanthogranulomatous inflammation was missed as a differential diagnosis.

Keywords: Histiocytes, oophoritis, xanthogranulomatous

How to cite this article:
Bindu SM, Mahajan MS. Xanthogranulomatous oophoritis: A case report with review of literature. Int J Health Allied Sci 2014;3:187-9

How to cite this URL:
Bindu SM, Mahajan MS. Xanthogranulomatous oophoritis: A case report with review of literature. Int J Health Allied Sci [serial online] 2014 [cited 2021 Mar 8];3:187-9. Available from: https://www.ijhas.in/text.asp?2014/3/3/187/138604

Introduction

The presence of a large number of lipid containing macrophages with an admixture of lymphocytes, plasma cells, and neutrophils is characteristic of xanthogranulomatous inflammation. Multinucleated giant cells may also be present. Xanthogranulomatous inflammation is a form of chronic inflammation that is destructive to the normal tissue of affected organs. ^[1] Most commonly affected organ is kidney, followed by gall bladder. ^[2] Other organs in which xanthogranulomatous inflammation has been reported are stomach, anorectal area, bone, urinary bladder, testis, epididymis and female genital tract. Only a few cases of xanthogranulomatous oophoritis have been reported till date. ^[3],[4],[5]

Case report

A 40-year-old female presented with abdominal pain and bleeding per vaginum since 8 days. On examination, there was a mass in the right pelvic region. Computed tomography scan showed multiloculated septate lesion in the right adenexa, suggestive of mucinous cystadenoma or mucinous carcinoma. Total abdominal hysterectomy with bilateral salpingo-oophorectomy was done. We received a specimen of uterus with one sided Fallopian tube ^{More Details} and ovary, along with a separate tubo-ovarian mass and a piece of omentum. Size of the mass was 7 cm × 5 cm × 5 cm; cut surface was partly solid, with a yellowish tinge and partly cystic [Figure 1]. Histologically, it showed ovarian stroma almost completely replaced with dense inflammatory cell infiltrate consisting of large number of foamy histiocytes, accompanied by lymphocytes and plasma cells and plenty of necrosis. There was no evidence of malignancy in the sections studied from the tubo-ovarian mass [Figure 2].

Figure 1: Gross appearance showing partly cystic and partly solid yellowish mass

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Figure 2: Microscopic picture showing replacement of ovarian stroma by foamy histiocytes

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Discussion

Xanthogranulomatous oophoritis is a rare form of chronic oophoritis. ^[6] The inflammation most often affects the endometrium in the female genital tract, but involvement of the vagina, cervix, fallopian tube and ovary may also occur. ^[7] Only a few cases of xanthogranulomatous oophoritis have been reported from India. Only 14 cases of xanthogranulomatous inflammation involving the ovary have been reported until date. Kunakemakorn was the first to describe xanthogranulomatous inflammation of serosa of the uterus, left fallopian tube and ovary in his report of inflammatory pseudotumour in the pelvis in 1976. ^[8] The exact pathogenesis of xanthogranulomatous inflammation is still unclear. Proposed etiologic factors include chronic bacterial infections, ineffective antibiotic therapy, ineffective clearance of bacteria by phagocytes, abnormality in macrophage, Gram-negative or anerobic bacteria as in genitourinary tract infections, foreign material such as retained suture material ^[9] and long standing pelvic inflammatory disease. ^[10],[11] Bacteria like Bacteroides fragilis, Escherichia ^{More Details} coli, Staphylococcus aureus, Salmonella ^{More Details} typhi can be considered in the pathogenesis of xanthogranulomatous oophoritis. Punia et al. have reported a case of xanthogranulomatous oophoritis and salpingitis as a late sequelae of inadequately treated staphylococcal pelvic inflammatory disease. ^[12],[13] Shukla et al. have reported a case of xanthogranulomatous oophoritis associated with primary infertility and endometriosis. ^[14] Premature ovarian failure as a rare sequelae of xanthogranulomatous inflammation has also been reported. ^[15] Cases of xanthogranulomatous inflammation of ovary with ovarian hemangioma, ^[16] secondary to diverticulitis, ^[17] as an unusual cause of tubo-ovarian abscess, ^[18] association with endometriosis and uterine leiomyoma, ^[19],[20] association with diabetes mellitis, ^[21] secondary to talcum powder, ^[22] presenting as an unusual complication of typhoid ^[23] and following uterine artery embolization ^[24] have been reported.

The average age of patients with affected ovaries is 31 years ^[25],[26] and the youngest case reported was of 18 years. The clinical presentations include pain in abdomen, fever, abdominal mass, menorrhagia, anemia, and anorexia. Gynecological examination reveals adenexal mass with tenderness. Laboratory tests show elevated erythrocyte sedimentation rate and raised white blood cell count. Radiological findings of xanthogranulomatous oophoritis may simulate a malignant ovarian neoplasm. Grossly, the involved ovary is replaced by a solid, yellow, well-circumscribed, lobulated mass, sometimes involving adjacent organs, thus simulating malignancy. This mass can be occasionally cystic due to liquefactive necrosis. Histologically, foamy histiocytes (xanthoma cells), and chronic inflammatory cells are consistently observed. Xanthoma cells are histiocytes with abundant lipid laden cytoplasm having vacuolated appearance. It is aggregates of such foam cells, which are responsible for the yellow color observed on gross examination. ^[27] The emergence of foam cells may be attributed to the following factors:

Inefficient or inappropriate antibiotics applied in the early phase of infection that resulted in ineffective control of bacterial multiplication.
Presence of a lipid metabolic disorder that induces hyperlipidemia and the foam cells are formed when the lipid deposited is phagocytosed by phagocytes.
The application of intrauterine contraceptive devices or drugs, ^[28] involvement of adjacent organs and pelvic peritoneum results in adhesions, hence arousing the suspicion of malignancy.

The present case is consistent with the clinical findings of other reported cases of xanthogranulomatous oophoritis. We received a specimen of abdominal hysterectomy with bilateral ovaries and tubo-ovarian mass with a piece of omentum, which suggests strong clinical suspicion of malignancy. Gross and histologic findings were also characteristic. The treatment of choice for xanthogranulomatous oophoritis is oophorectomy. Though antibiotic therapy has been attempted, it has been of little use in reducing the size of the mass. Since this entity is usually associated with pelvic inflammatory disease, endometriosis, intrauterine death, these patients should be followed-up closely for early diagnosis. Awareness of this entity is vital in order to prevent misdiagnosis of malignancy and avoid radical surgery.

Conclusion

Xanthogranulomatous inflammation of the ovary is an uncommon entity. The clinical and radiological features of this entity may mimic an ovarian neoplasm, so it must be considered in the differential diagnosis of ovarian tumors. Second, most of the patients using intrauterine contraceptive devices and those of pelvic inflammatory disease and endometriosis need close follow-up as they are prone for xanthogranulomatous oophoritis; so that they are diagnosed early and radical surgery is avoided.

Acknowledgment

The authors are thankful to the authorities of MGM Medical College, Aurangabad, Maharashtra, India for providing necessary facilities for the concerned case study.

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