OHSS, though a rare complication of ovarian stimulation, but if not detected early and adequately managed can lead to life threatening consequences.

Objective: To report a case of a variant of hypogonadotropichypogonadism with severe OHSS and to discuss the risk factors and strategies of prevention.

Case report: Here, we present a case of hypogonadotropichypogonadism with polycystic ovarian morphology. She was a thin built woman with eight years of infertility. Her AMH value was on a higher side and on ovulation induction. She developed severe late onset OHSS on day 11 of the HCG trigger which was successfully managed.

Conclusion: While doing ovulation induction in patients with hypogonadotropichypogonadism, it should be kept in mind that there exists a variant with polycystic ovarian morphology (PCOM), who might show hyper–response to gonadotropin stimulation. These are the patients who are at high risk of OHSS and need more frequent monitoring. Serum AMH and polycystic morphology on stimulation can help suspect OHSS at an early stage. Luteal coasting or use of progesterone’s in place of HCG for luteal phase support can help in decreasing the risk of severe OHSS.