Endosalpingiosis is characterized by the presence of ectopic, benign glands with a fallopian tube-like ciliated epithelium. Florid cystic endosalpingiosis (FCE) is a rare type of endosalpingiosis and presents with tumor-like lesions. In general, FCE has no specific clinical features. In this case, extensive pelvic multiple Müllerian cysts were first observed and removed during the patient's second cesarean section. Lesions relapsed after a year. Therefore, the patient underwent total hysterectomy and bilateral salpingectomy; pathology revealed that the patient had FCE. According to imaging studies during the follow up, recurrent and progressive multiple pelvic and extra-pelvic cysts were observed. The patient had no obvious symptoms, and the results of her laboratory tests were within normal limits. Ultrasound-guided aspiration and lauromacrogol sclerotherapy were performed, and in the past year, the cysts have stabilized without progression. This is the first reported case of recurrent FCE after total hysterectomy and bilateral salpingectomy with a 5-year follow up. A literature review and novel ideas for diagnosing and managing FCE based on this case are also presented.
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