Aim: Ovarian hyperstimulation syndrome (OHSS) is a potentially life-threatening, iatrogenic complication of assisted reproduction and has been associated with poor in vitro fertilization outcome. The aim of the present study was to evaluate the pregnancy rate and outcome following severe OHSS, at a single center over a three-year period. Methods: The incidence of severe OHSS at the IVF Center, National University of Singapore, in Singapore, was 4% (48 cases over 1200 cycles) during the period of 1997-2000. The present retrospective study compared 48 cases of severe OHSS to 144 age-matched, contemporaneous controls without OHSS. Results: The total gonadotropin required for severe OHSS group was found to be lower than for that of controls (2664.06 ± 768.29 IU vs 3349.58 ± 2003.73 IU), although duration of stimulation was similar. The OHSS group was associated with a fivefold increase (OR 5.293, 95% CI: 2.116-13.238) in pregnancy rate compared to controls (87.5% vs 56.9%; P 0.05) and miscarriage rates (14% vs 7.3%; P > 0.05) between the groups. Conclusion: Severe OHSS at our center were mostly late onset. The pregnancy rate was significantly higher, but multiple pregnancy and miscarriage rates were not significantly increased when compared to the age-matched contemporaneous controls. (Reprod Med Biol 2005; 4: 207-211).