Abstract： Objective To summarize and share experiences of diagnosis and treatment of septate uterus with double cervix and longitudinal vaginal septum. Methods Original data was retrieved from electronic medical record database of Peking Union Medical College Hospital, specific malformations were confirmed according to the description of operation records, and patients concerned were followedup by outpatient medical records and telephone interview.Results All 10 patients complained of infertility, and underwent resection of vaginal septum and laparo-hysteroscopic surgery, with only one exception of open operation for uterine orthopedic.Three patients had spontaneous pregnancy and full-term cesarean section delivery, interval between pregnancy and previous surgery being 2~3.5 years.Conclusion Septate uterus with double cervix and longitudinal vaginal septum is a complex mullerian anomaly in patients with infertility. Hysteroscopic resection of uterine septum is the priority recommendation,keeping double cervix intact.