OBJECTIVETo report a successfully managed case of extensive intracranial sinus thrombosis that occurred during an IVF cycle, and to review the literature.DESIGNCase report and review of literature.SETTINGUniversity-affiliated teaching hospital.PATIENT(S)A 38-year-old nulliparous woman who developed severe ovarian hyperstimulation syndrome on luteal day (LD) 5 during an IVF cycle. Hemoconcentration was corrected, ascitis drained, and heparin prophylaxis started. On LD7, the patient complained of severe headache and neck pain. A magnetic resonance imaging (MRI) study of the head showed extensive cortical vein and dural sinus thrombosis, including the superior sagittal sinus and transverse sinuses.INTERVENTION(S)The patient was fully heparinized using low-molecular-weight heparin. On LD15 she was discharged home on warfarin, after confirming a negative pregnancy test. Complete thrombophilia work-up was negative.MAIN OUTCOME MEASURE(S)Clinical outcome.RESULT(S)Repeat MRI 2 months later revealed patent superior sagittal sinus and transverse sinuses, unremarkable cortical veins, and no evidence of flow obstruction. The patient was asymptomatic, with no neurologic sequelae.CONCLUSION(S)Extensive intracranial sinus thrombosis in women undergoing IVF may present with minimal symptoms and can occur in the absence of pregnancy and thrombophilia and despite heparin prophylaxis and correction of hemoconcentration. Medical management was successful and left the patient with no neurologic sequelae.