Background: Cesarean scar syndrome results from a postoperative defect of the uterine isthmus, also known as an isthmocele. Patients present with gynecological symptoms, such as abnormal genital bleeding or infertility, after cesarean delivery. Although the cesarean rate is increasing worldwide, this syndrome is not widely known. Case presentation: A 43-year-old G2P1 Japanese woman with atypical cesarean scar syndrome had a 3-year history of secondary infertility and postmenstrual brown discharge. Laparoscopic and hysteroscopic exploration revealed a cesarean scar defect connected to a small cavity in the myometrium: this was not an endometrial cavity or a uterine diverticulum. After endoscopic excision of the cavity, the brown discharge resolved, and the patient achieved ongoing pregnancy on her third attempt at intrauterine insemination. Conclusion: Consensus is still lacking regarding the diagnosis and treatment of cesarean scar defect. However, the gynecologists should be aware that cesarean scar syndrome can have scar defects forming cavities of unusual shapes and features. Surgical correction of these defects will often improve postmenstrual bleeding and subfertility in these cases.
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