ABSTRACT
INTRODUCTION AND IMPORTANCE: Xanthomatous inflammation is a rare chronic inflammatory condition typically affecting organs such as the kidney and gallbladder. Its occurrence in the female genital tract, particularly in the ovaries and fallopian tubes, is exceptionally rare and sparsely documented. CASE PRESENTATION: We report a unique case of xanthomatous inflammation involving the fallopian tube and ovary, characterized by the presence of hobnail cells and apocrine metaplasia. This represents the first documented instance in medical literature. A 55-year-old woman presented with pelvic masses, initially raising suspicion of more common conditions such as ovarian neoplasms or tuberculosis. CLINICAL DISCUSSION: Xanthomatous salpingo-oophoritis (XSO) often presents with symptoms resembling ovarian tumors or infectious diseases, posing challenges in diagnosis. Accurate preoperative identification is essential to avoid unnecessary radical surgeries and optimize patient management. CONCLUSION: This case highlights the importance of considering xanthomatous inflammation in the differential diagnosis of ovarian and tubal lesions, especially when typical symptoms of pelvic masses are present. Recognizing this rare inflammatory condition can prevent overtreatment and guide appropriate therapeutic strategies.