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A 28 years old multigravid admitted to our maternal fetal medicine unit because of a second trimester loss of pregnancy due to preterm labor at 28 week. Her obstetrical history was uneventful. Ultrasonographic examination revealed a unicornuate uterus with rudimentary horn. Hyperhomocysteinemia caused by MTHFR heterozy gosity and decrease in Protein S levels were found by blood analysis. Low dose aspirine and LWM Heparin were started 6 weeks bef ore conception. Follow-up of the pregnancy in the functional unicornus resulted in a healthy baby delivered by cesarean section at 37 week due to chronic intrauterine hypoxia and uterine anomaly.