ObjectiveDescribe outcomes of open fetal surgery for myelomeningocele (MMC) repair in two Brazilian hospitals and the impact of surgical experience on outcome.DesignRetrospective cohort study.SettingSao Paulo, Brazil.Population237 pregnant women carrying a fetus with an open spinal defect.MethodsSurgical details, and maternal and fetal outcomes collected from all patients.Main outcome measuresAnalysis of surgical and perinatal outcome parameters.ResultsTotal surgical time was 119 ± 7.6 minutes. Preterm labour occurred in 24.2%, premature rupture of membranes in 26.7%, placental abruption in 0.8%, need for a blood transfusion at delivery in 2.1%, and dehiscence at the repair site in 2.5%. Reversal of hindbrain herniation at birth occurred in 71.4%. There were no maternal deaths or severe maternal morbidities. The failure rate with the patient anaesthetised was 0.42% and perinatal mortality was 2.1% (three intrauterine demises and two neonatal deaths). Comparing results from our study in the first 3 years with the last 3 years demonstrated improvement in the total surgical time (121.2 ± 6.4 versus 118.5 ± 8.2 minutes, P = 0.005) and an increase in reversal of hindbrain herniation at birth (64.0 versus 77.1%, P = 0.042).ConclusionOur open fetal surgical approach for MMC was effective and results were comparable to past studies. Improvements in surgical performance and perinatal outcome increased as the surgical team became more familiar with the procedure.FundingThe study was funded solely by institutional funds.Tweetable abstractBrazilian experience of in utero open surgery for myelomeningocele repair.