BACKGROUND An isthmocele is a scar defect in the uterine wall due to a cesarean section. Its prevalence is unknown, but it has been associated with secondary infertility. Here, we present a case where the patient suffered from an isthmocele that was associated with persistent hydrometra, which developed during in vitro fertilization. CASE SUMMARY The patient underwent hysteroscopic surgery, which successfully resolved the isthmocele as well as the hydrometra. Afterward, two high-quality, euploid embryos, determined by morphological assessment and pre-implantation genetic diagnostic testing, were transferred. This resulted in uterine pregnancy, as determined by serum β-human chorionic gonadotropin levels on day 14 (180 mU/mL) and ultrasound-confirmed presence of a gestational sac with a positive embryocardia at week 6. The pregnancy reached 36 wk without any complications, and the product was born in good health. We report a successful isthmocele treatment in a patient with secondary infertility, in which the isthmocele was the cause of persistent hydrometra. CONCLUSION Hydrometra caused by secondary cesarean is an infertility factor, which can be corrected by hysteroscopy plus ablation of the isthmocele.
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