Uterine endosalpingiosis: Case report and review of the literature

Abstract

Objective: Endosalpingiosis rarely occurs in the uterus. We report a case and review literature to explore its current clinical diagnosis and treatment. Case report: A 31-year-old woman was admitted to the hospital for suspected uterine leiomyoma with cystic degeneration based on ultrasound and magnetic resonance imaging and surgical treatment. Laparoscopy revealed a large cystic mass protruding from the posterior uterine wall. The mass was resected, and a histologic examination showed that the cyst wall was lined with benign fallopian tube–type ciliated epithelium surrounded by normal myometrium, consistent with the diagnosis of endosalpingiosis of the uterus (also known as a Müllerian cyst). Currently, there are 18 cases (including ours) in the literature. Of these, two had a uterine malignancy, one endometrioid endometrial carcinoma, and another cervical adenocarcinoma. The age at diagnosis varied from 31 to 73, with a mean of 47 ± 8. The typical clinical manifestations were a palpable low abdominal mass, abnormal menstrual bleeding, and pelvic pain. Overall, 75% (12 of 16) of patients underwent a total hysterectomy, while 62.5% (10 in 16) had a concomitant bilateral salpingo-oophorectomy for nonmalignancy. No recurrence was reported. Conclusion: We describe the youngest patient with tumor-like uterine endosalpingiosis. The preoperative diagnosis is challenging because of its rarity. Most patients had a hysterectomy with castration, which may have resulted in overtreatment. Awareness of this lesion is necessary for the differential diagnosis of uterine and adnexal tumors. Review of relevant literature has shown a relationship between endosalpingiosis progression and cancer development, indicating an uncertain and complicated pathology. Keywords: Endosalpingiosis, Müllerian cyst, Müllerianosis, Uterus