Endometrial malacoplakia is an extremely rare condition, as demonstrated by the paucity of cases reported in the international literature. The etiopathogenesis of the disease is far from being completely understood, but recent evidence agrees with the presence of defective macrophage phagolysosomal activity [
1]. Several studies reported a correlation between malacoplakia and immunodeficiency conditions, such as primary immunodeficiency, steroid administration, AIDS and diabetes [
2]. The defect in the phagolysosomal activity is believed to be a consequence of immunodeficiency, leading to incomplete bacterial digestion and to Michaelis-Gutmann bodies formation. These bodies are considered as a pathognomonic microscopical sign of malacoplakia, but they are not strictly necessary for the diagnosis, as they may not be seen in early stages of the disease. The case presented in this report developed endometrial malacoplakia after an abortion and subsequent uterine curettage due to retention of placental rests. On the basis of the clinical history, we might suppose a bacterial contamination leading to the development of the disease, but in the absence of a known condition of immunodeficiency. Moreover, due to placental rests retention, we might hypothesize a role played by trophoblast. In fact, cytokines released by trophoblast cells have been reported to be involved, among various functions, in immunoregulation and maternal-fetal tolerance during pregnancy [
3,
4]. The clinical presentation of endometrial malacoplakia is characterized by post-menopausal bleeding, abnormal uterine bleeding in a menstruating woman or suspicious uterine mass [
5‐
7]. Our patient presented with abundant vaginal sero-hematic secretions and received a further curettage, in order to establish a possible placental rests retention, but the histological examination revealed the microscopical features of endometrial malacoplakia and the absence of chorionic rests. Due to its clinical presentation and endoscopical appearance, uterine malacoplakia requires a careful differential diagnosis, in order to rule out other inflammatory processes and uterine neoplasms. The therapeutic approach to malacoplakia is based on surgical and medical options, but there are no established guidelines. Surgical intervention varies in accordance to site and extension of the disease. Antibiotic therapy is prevalently based on drugs capable of reaching high concentrations inside macrophages, such as quinolones or trimethoprim-sulfamethoxazole [
8,
9]. Currently, antibiotics, combined with surgery, provide the best therapeutic protocol. A novel protocol, including antibiotics, bethanechol and ascorbic acid, has been recently used in the treatment of cerebral malacoplakia [
10]. In the case herein reported the patient was treated with uterine curettage, combined with quinolones antibiotic therapy, obtaining a rapid clinical improvement, with satisfactory disappearance of symptoms, as also reported by the patient. Even though management of this rare condition is very challenging, the prognosis is commonly good. Nevertheless, recurrence, as in the case we reported herein, and complications may frequently occur throughout the years.