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Objective: To describe the management of bicornuate uterus with ipsilateral renal agenesis. Case Report: Two patients were diagnosed with bicornuate uterus and ipsilateral renal agenesis; one of which was identified as bicornuate unicollis. Both patients were successfully treated with metroplasty. In one patient, the central myometrium was found to extend to the level of the internal cervical os, suggesting bicornuate unicollis. Dysmenorrhea decreases during menstrual periods and there is no restriction on childbearing. Conclusion: Bicornuate uterus carries the potential risks of back flow of menstrual secretion and ascending infection. Metroplasty is a valuable treatment for bicornuate uterus with a single cervix and vagina, and also prevents the back flow of menstruation which would otherwise progress to pelvic endometriosis.