Xanthomatous salpingo-oophoritis accompanied by hobnail cell and apocrine metaplasia: The first case report in the literature.

A 55-year-old nulliparous woman presented to our gynecology clinic with a three-month history of progressively worsening lower abdominal pain localized to the left lower quadrant, accompanied by occasional low-grade fever. Her last menstrual period was five years ago. She also reported constant abnormal vaginal bleeding over the past six months, despite multiple courses of oral hormonal therapy. During the physical examination, tenderness and mild guarding were noted in the left lower abdomen, with no palpable masses. Pelvic examination revealed a slightly enlarged globular firm uterus and a notable amount of white vaginal discharge. Laboratory analysis showed a hemoglobin level of 7.8 g/dL, an iron level of 23 μg/dL, a total leukocyte count of 12.3 × 10^9/L, a C-reactive protein level (CRP) of 28.9 mg/dL, and a urine routine with 10–15 pus cells per high power field. Transvaginal ultrasound (TVUS) revealed a tubo-ovarian mass lesion in the left adnexa, measuring approximately 5.5 cm × 4 cm. The uterus had abnormal measurements, and free fluid in the cul-de-sac suggested an inflammatory condition. Given the presence of fever, elevated CRP, leukocytosis, and white vaginal discharge, an initial diagnosis of pelvic inflammatory disease (PID) was considered likely. Consequently, oral antibiotics were prescribed. Despite this, the patient showed no clinical improvement, necessitating a switch to intravenous antibiotics. The persistence of symptoms and concerning ultrasound findings warranted consideration of malignancy in the differential diagnosis. Given the acute presentation and risk of worsening infection, a decision for laparotomy was made to obtain a definitive diagnosis and address the patient's symptoms. The choice of laparotomy over laparoscopic surgery was influenced by the suspected extensive adhesions and the need for a thorough exploration, which is often more feasible through an open approach. During the surgery, a well-circumscribed, solid, and lobulated mass was identified in the left adnexa. The mass was carefully excised and sent for histopathological examination. The procedure was performed under general anesthesia, and the patient was positioned in a lithotomy position. A lower midline incision was made to gain access to the pelvic cavity. The mass was found to be adherent to the surrounding structures but was successfully resected without intraoperative complications. The operative field was thoroughly inspected to ensure complete removal and hemostasis. The excised specimen was immediately sent for pathological evaluation. Gross pathology revealed a well-circumscribed, solid, lobulated mass measuring 9 cm × 5 cm × 4 cm in the left adnexa. Upon sectioning, solid, yellowish areas were observed [ Fig. 1 ]. Fig. 1 A gross image of the specimen shows fragments mass. Upon sectioning, solid, yellowish, areas are observed (circles). Fig. 1 A gross image of the specimen shows fragments mass. Upon sectioning, solid, yellowish, areas are observed (circles). Microscopic examination confirmed marked inflammation, foamy macrophages with lipid droplets consistent with xanthomatous change, and chronic inflammatory infiltrates [ Fig. 2 ]. Histological sections exhibited areas of hobnail cell metaplasia and apocrine metaplasia [ Fig. 3 ], with apocrine cells demonstrating periodic acid-Schiff (PAS) positivity [ Fig. 4 ]. Based on these findings, a diagnosis of xanthomatous salpingo-oophoritis was made. Fig. 2 Hematoxylin and eosin stain (H&E) images (A-D) depict microscopic views of the mass. Foamy macrophages with acute and chronic inflammatory infiltrates are evident at 40×, 100×, 200×, and 400× magnification. Fig. 2 Fig. 3 Hematoxylin and eosin stain (H&E) images (A-D) reveal different cellular changes: (A and B) Areas of hobnail cell metaplasia characterized by elongated and hyperchromatic nuclei, resembling a hobnail-shaped appearance (arrows) at 200× magnification. (C and D) Scattered areas of apocrine metaplasia, characterized by epithelial cells with eosinophilic cytoplasm and prominent apical cytoplasmic snouts, also at 200× magnification. Fig. 3 Fig. 4 Periodic acid-Schiff (PAS) stain demonstrates the positivity of apocrine cells at 200× magnification. Fig. 4 Hematoxylin and eosin stain (H&E) images (A-D) depict microscopic views of the mass. Foamy macrophages with acute and chronic inflammatory infiltrates are evident at 40×, 100×, 200×, and 400× magnification. Hematoxylin and eosin stain (H&E) images (A-D) reveal different cellular changes: (A and B) Areas of hobnail cell metaplasia characterized by elongated and hyperchromatic nuclei, resembling a hobnail-shaped appearance (arrows) at 200× magnification. (C and D) Scattered areas of apocrine metaplasia, characterized by epithelial cells with eosinophilic cytoplasm and prominent apical cytoplasmic snouts, also at 200× magnification. Periodic acid-Schiff (PAS) stain demonstrates the positivity of apocrine cells at 200× magnification. Postoperatively, the patient's recovery was uneventful, and follow-up assessments at four months revealed no evidence of recurrence.

Diana Nouh: study design, data collections, data analysis, and writing. Farah Issa: study design, data analysis, and writing. Eman Alwaraa study design, data analysis, and writing. Louai Hasan: performed surgery. Moatasem Hussein Al-janabi: study design, data collections, data analysis, writing, and reviewing the manuscript.

Written informed consent was obtained from the patient for the publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

No ethical approval was required for this case report as it involves no experimental interventions.

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Not applicable.

Moatasem Hussein Al-janabi.

Xanthomatous Salpingo-Oophoritis represents a rare manifestation of chronic inflammation within the genitourinary system. Despite sharing histopathological characteristics with inflammatory processes in other organs such as the gallbladder and kidney, the exact etiology of XSO remains elusive. Our presentation of a unique case of xanthomatous salpingo-oophoritis accompanied by hobnail cell and apocrine metaplasia highlights the necessity of considering rare pathological entities in the differential diagnosis of ovarian and tubal lesions. This case contributes to the existing literature by providing the first documented instance of these combined histopathological features in XSO, emphasizing the complexity of diagnosing such conditions. These findings suggest that clinicians should be aware of the potential for rare inflammatory conditions to mimic more common gynecological malignancies or infections, which can significantly impact the management strategy. Early and accurate diagnosis through comprehensive histopathological examination can prevent unnecessary radical surgeries and associated morbidities. Future research should focus on elucidating the etiopathogenesis of XSO and exploring minimally invasive approaches for its management, which could further refine treatment protocols and improve patient outcomes.

Xanthomatous inflammation represents an infrequent chronic inflammatory process, with its occurrence in the female genital tract being rare [ 8 ]. The exact etiology of XSO remains elusive, with proposed hypotheses including abnormalities in lipid metabolism, antibiotic treatment, cervical stenosis, intrauterine contraceptive device usage, endometriosis, and infections with various organisms [ 9 , 10 ]. Interestingly, no association has been established between XSO and malignancy or radiotherapy [ 11 ]. Patients with XSO typically present with symptoms resembling pelvic inflammatory disease, such as pelvic pain, fever, abnormal vaginal discharge, spotting between periods, dysmenorrhea, pain during ovulation, painful intercourse, lower back pain, frequent urination, nausea, vomiting, and potential infertility [ 1 , 6 ]. In the presented case, the patient exhibited lower abdominal pain, a low-grade fever, and white vaginal discharge before the diagnosis of XSO, the etiology of which remained undetermined. Differential diagnosis includes non-neoplastic and neoplastic conditions, with histopathological examination being essential for accurate diagnosis. Treatment options vary based on the severity of the condition and may involve antibiotics, hospitalization, and surgery. Surgical interventions, such as laparoscopy with abscess drainage or total abdominal hysterectomy-bilateral salpingo-oophorectomy, may be necessary in cases resistant to drug treatment [ 12 ]. In the presented case, following multiple unsuccessful antibiotic courses, surgical intervention was recommended, resulting in a successful outcome with no evidence of recurrence at the six-month follow-up. Hobnail cell metaplasia, characterized by epithelial cells with elongated and hyperchromatic nuclei, typically presents in various gynecological malignancies, particularly ovarian clear cell carcinoma [ 13 ]. However, its occurrence in the context of XSO is exceptionally rare and has not been previously reported in the medical literature. The significance of hobnail cell metaplasia in the pathogenesis of XSO remains uncertain and warrants further investigation. Apocrine metaplasia, characterized by the presence of epithelial cells with eosinophilic cytoplasm and prominent apical cytoplasmic snouts, is a common finding in benign breast lesions [ 14 ], but is rarely reported in ovarian pathology. Its association with XSO adds another layer of complexity to the histopathological spectrum of this rare condition. The coexistence of XSO with hobnail cells and apocrine metaplasia in our case underscores the importance of thorough histopathological examination in the diagnosis of ovarian and tubal lesions. While XSO is typically considered a benign inflammatory condition, the presence of additional metaplastic changes may raise concerns for potential neoplastic transformation or concurrent malignancy. Therefore, careful clinicopathological correlation is essential for accurate diagnosis and appropriate management. The implications of these findings for clinical practice are significant. Recognizing xanthomatous salpingo-oophoritis (XSO) as a differential diagnosis in patients presenting with pelvic masses can prevent misdiagnosis and avoid unnecessary radical surgeries. Histopathological examination remains crucial for differentiating XSO from malignancies or other inflammatory conditions. Screening programs for the prevention of gynecological malignancies play a crucial role in the early identification and management of unusual cases like XSO, underscoring the importance of regular gynecological check-ups and vigilance in clinical practice [ 15 ]. Future research should aim to understand the etiopathogenesis of XSO better and investigate the role of minimally invasive surgical techniques in managing adnexal lesions. A minimally invasive approach, such as laparoscopy, has been shown to reduce patient morbidity and recovery time while providing adequate treatment for adnexal masses [ 16 ]. Further studies are warranted to evaluate the effectiveness and safety of these techniques in the context of rare inflammatory conditions like XSO.

Not commissioned, externally peer-reviewed.

Xanthogranulomatous inflammation is an uncommon cause of chronic inflammation, characterized by the accumulation of lipid-laden foamy macrophages intermixed with neutrophils, lymphocytes, and plasma cells, along with multinucleated giant cells, known as Touton giant cells [ 1 , 2 ]. This inflammatory process leads to the destruction of normal tissue within the affected organ [ 1 ]. It is primarily observed in organs such as the kidney and gall bladder but can also affect various organs including the stomach, anorectal area, bone, urinary bladder, testis, and epididymis [ 3 ]. However, its occurrence in the female genital tract is exceptionally rare, with only a handful of cases reported, usually involving the fallopian tube and ovaries [ 3 ]. Xanthomatous Salpingo-Oophoritis (XSO) exemplifies this pathological process, leading to functional impairment of the reproductive organs due to profound structural damage characterized by the accumulation of lipid-laden foamy macrophages within the fallopian tubes and ovaries [ 4 ]. XSO primarily affects females of reproductive age, typically ranging from 23 to 72 years old [ 5 ]. The resulting pelvic dysfunction and anatomical distortion contribute to symptoms such as chronic pelvic pain, infertility, fever, and abnormal bleeding [ 6 ], often resembling other gynecological disorders. Associations with pelvic inflammatory disease, intrauterine contraceptive devices, and extensive endometriosis have been observed, with proposed etiological factors including abnormalities in lipid metabolism, infections, ineffective antibiotic therapy, and impaired bacterial clearance by phagocytes [ 4 ]. Nonetheless, the precise etiology of XSO remains elusive [ 6 ]. While XSO is acknowledged as a rare condition, its correlation with other pathological features such as hobnail cell and apocrine metaplasia is scarcely documented in medical literature. In this report, we present a unique case of XSO accompanied by hobnail cell and apocrine metaplasia, which, to the best of our knowledge, represents the first reported instance in the medical literature. This case report has been reported in line with SCARE Criteria [ 7 ].

The authors have no conflicts of interest to declare.