Background: Thromboembolic complications, renal failure and severe respiratory failure were observed in 0.5% of the women treated for ovary hyperstimulation syndrome (OHSS). Case Report: A 38-year-old nulligravida underwent first ICSI-PESA cycle with obstructive azoospermia. Three fresh embryos on day 3 were transferred, cerebral vein thrombosis secondary to OHSS occurred at early gestation. This is one of the few reported cases that illustrates the delivery of healthy baby in patient of cerebral vein thrombosis secondary to OHSS. Conclusion: Several factors have been suggested to correlate with OHSS, and thrombosis is a severe and rare complication. Timely treatment is prone to a good prognosis.