Xanthogranulomatous Oophoritis: Sequelae of Pelvic Inflammatory Disease in a Diabetic Woman

Abstract

Background:

Xanthogranulomatous inflammation is an uncommon form of chronic granulomatous inflammation commonly reported in the kidneys and gallbladder. This inflammation rarely affects the female genital tract and usually involves the endometrium. Involvement of the ovaries is a rare phenomenon known as xanthogranulomatous oophoritis (XO).

Case:

A 45-year-old woman presented with left-sided abdominal pain and fever that she had for a duration of 3 months prior. Her clinical diagnosis was chronic pelvic inflammatory disease, and imaging suggested a differential diagnosis of an ovarian malignancy.

Results:

Microscopic examination of her lesion showed lipid-laden foamy macrophages intermixed with lymphocytes, giant cells, and plasma cells, all of which were consistent with XO with organized abscess formation.

Conclusions:

Xanthogranulomatous inflammation of the ovary is an uncommon entity. The clinical and radiologic features of this entity can mimic an ovarian neoplasm, so XO must be considered in the differential diagnosis of ovarian lesions.

Introduction

Xanthogranulomatous inflammation is an uncommon, nonneoplastic chronic process in which the tissue of the affected organ is destroyed and is replaced by a striking cellular infiltrate of foamy histiocytes admixed with multinucleated giant cells, plasma cells, fibroblasts, neutrophils, and foci of necrosis.¹ This inflammation is an established histopathologic entity with pathognomonic imaging findings in the kidney and gallbladder, but rarely involves the female genital tract, and most often affects the endometrium. Xanthogranulomatous oophoritis (XO) is an uncommon form of chronic inflammation affecting the ovaries with <20 cases reported so far in the English literature.² XO is difficult to diagnose preoperatively, as it can present with nonspecific symptoms of abdominal pain and a vague adnexal mass mimicking an ovarian tumor and/or a tubo-ovarian abscess.

Case

A 45-year-old woman attended the outpatient department, with complaints of left-sided abdominal pain, intermittent fever, and vomiting for a duration of 3 months, with associated symptoms of a urinary tract infection. Her menstrual history was unremarkable, and she had no abnormal vaginal discharge. She was a known case of type 2 diabetes mellitus and had been taking insulin for the past 2 years. A physical examination revealed left iliac fossa tenderness. A vague mass was palpable in the left fornix on vaginal examination. The cervix and vagina were healthy with a normal-size uterus. A basic laboratory work-up yielded normal results, including her cancer antigen–125 level, except for an elevated HbA1c level at 12%. A polymerase chain reaction test (PCR) for tuberculosis was negative. A Papanicolaou smear showed inflammatory cells with no evidence of an intraepithelial neoplasm.

A pelvic ultrasound (US) revealed a complex cystic left adnexal mass and computed tomography (CT) of her abdomen was performed for further characterization. CT showed a multiloculated cystic lesion in the left adnexa with thick, enhancing internal septations. The right ovary was unremarkable, and there were no ascites or peritoneal deposits. The possibility of a left tubo-ovarian abscess or a cystic ovarian neoplasm was considered according to the imaging (Fig 1).

FIG. 1.

(A, B) Computed tomography scans of the patient's abdomen with contrast shows complex solid cystic left adnexal mass (arrows).

She underwent a laparotomy with findings of a left ovarian mass containing purulent material. There were no omental deposits or ascites. Her uterus and contralateral tube and ovary were normal.

A left oophorectomy was performed, followed by peritoneal lavage. Pathologic examination revealed that she had multiloculated cystic mass with multiple yellowish intramural nodules, dark serosanguinous fluid, and necrotic debris.

Results

Microscopic examination showed lipid-laden foamy macrophages intermixed with lymphocytes, giant cells, and plasma cells, all of which were consistent with XO with organized abscess formation (Fig. 2).

FIG. 2.

Photomicrograph shows characteristic foamy macrophages, neutrophils, lymphocytes, and occasional plasma cells (hematoxylin and eosin stain at 400 × )

Discussion

Xanthogranulomatous inflammation of the female genital tract is uncommon and often limited to the endometrium. Only a few cases involving the ovary, vagina, cervix, and/or fallopian tubes have been reported. XO is a rare form of chronic oophoritis with the average age of patients being 31; the youngest documented case was in a 2-year-old girl. The etiology is unknown but reported risk factors include recurrent pelvic inflammatory disease (PID), inappropriate antibiotics therapy, use of intrauterine devices (IUDs) for contraception, diabetes mellitus, abnormality in lipid metabolism, endometriosis, and leiomyoma.³ Overall, the infection theory is the most accepted, which is supported by clinical evidence of infection and growth of bacteria such as Escherichia coli, Bacteroides fragilis, and Proteus vulgaris from the affected tissue in cultures.⁴

The clinical presentation is variable and includes lower abdominal pain, fever, an abdominal mass, menorrhagia, anemia, and anorexia. Gynecologic examination can show signs of PID such as cervical-motion tenderness and a palpable adnexal mass. US is the investigation of choice, which often shows complex cystic or, rarely, solid adnexal mass but cannot differentiate between an ovarian neoplasm and a tubo-ovarian abscess. CT can be performed to assess the extent of the mass; show if there is any solid enhancement to suggest malignancy; or reveal the presence of ascites, omental deposits, and/or lymphadenopathy. Magnetic resonance imaging findings in xanthogranulomatous oophoritis cases are not very well-described in the published literature, and one case report concluded that nonenhancing intramural nodules in the thickened wall of an ovarian cystic mass could be a unique feature of XO.⁵

On gross pathology, 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. Foamy histiocytes (xanthoma cells) and chronic inflammatory cells are consistently observed and are diagnostic of XO. Xanthoma cells are histiocytes with abundant lipid-laden cytoplasm having a vacuolated appearance. Aggregates of such foam cells are responsible for the yellow color observed on gross examination.⁶

Management often involves exploratory laparotomy as the preoperative diagnosis is indeterminate, with the differential diagnosis of a tubo-ovarian abscess and/or an ovarian malignancy. Treatment of choice is oophorectomy with salpingectomy if the fallopian tube is affected. Surgery can be difficult due to involvement of adjacent structures from chronicity and adhesions. Adequate peritoneal lavage and a careful search for omental deposits and lymph nodes are recommended to exclude an ovarian malignancy. Patient with risk factors, such as recurrent PID, endometritis, diabetes, and IUD use should be followed-up because of their close association with XO.^7,8

Conclusions

Clinical diagnosis in the current patient was in favor of chronic PID with a tubo-ovarian abscess. However, a possibility of an ovarian neoplasm was suggested on imaging. A high index of suspicion is essential to consider a preoperative diagnosis of XO, especially in patients with risk factors such diabetes, to avoid radical surgery. This case is of interest due to its rarity and XO's potential to mimic a tubo-ovarian abscess and/or an ovarian tumor.

Footnotes

Acknowledgment

The authors thank Dr. Kalaivani of the pathology department of SRM University College of Medicine & Health Sciences for her contribution to this article.

Author Disclosure Statement

The authors declare that there are no conflicts of interest.

References

Raj

, Jagadeesha

, Naveen

, Ramachandra

. Xanthogranulomatous oophoritis: Pathologic findings with clinical correlation. J Indian Med Assoc, 2012; 110:653.

Zhang

, Dong

, Zhang

, Desouki

, Zhao

. Xanthogranulomatous inflammation of the female genital tract: Report of three cases. J Cancer, 2012; 3:100.

Son

, Raetskaya-Solntseva

, Tirman

, Waters

, Kelly

. Xanthogranulomatous oophoritis presenting as an adnexal mass and bowel obstruction: A case report. J Reprod Med, 2015; 60(5–6):273.

Elahi

, Nigam

, Pujani

, Batra

. Xanthogranulomatous oophoritis mimicking malignancy: A diagnostic dilemma. BMJ Case Rep, 2015; 2015:bcr2015210642.

Jung

, Lee

, Han

, Hahn

. Xanthogranulomatous oophoritis: MR imaging findings with pathologic correlation. AJR Am J Roentgenol, 2002; 178:749.

Naik

, Madiwale

, Vaideeswar

. Xanthogranulomatous oophoritis—a case report. Indian J Pathol Microbiol, 1999; 42:89.

Altanis

, Raweily

, Katesmark

. Xanthogranulomatous endometritis and oophoritis secondary to diverticulitis: A rare cause of postmenopausal bleeding. J Obstet Gynaecol, 2007; 27:746.

Abeysundara

, Padumadasa

, Tissera

, Wijesinghe

. Xanthogranulomatous salpingitis and oophoritis associated with endometriosis and uterine leiomyoma presenting as intestinal obstruction. J Obstet Gynaecol Res, 2012; 38:1115.