Context Ovarian hyperstimulation syndrome (OHSS) is a potentially life threatening complication following assisted reproductive technology (ART). The incidence of severe cases is 1-5%. The pathogenesis is such that artificial stimulation of the ovaries leads to an increased expression of vascular endothelial growth factor (VEGF). This growth factor binds to its corresponding receptor, (VEGF2), resulting in increased vascular capillary permeability. The subsequent endothelial injury and vasodilation associated with release of pro inflammatory mediators leads to a fluid shift from the intravascular compartments to the third space. Hence, third space fluid loss associated with circulatory dysfunction is a well appreciated aetiology of the syndrome; often presenting with clinical symptoms of oliguria, ascites and pulmonary effusion. Objective To present a rare complication of acute hepatic dysfunction secondary to early onset severe ovarian hyperstimulation syndrome. Methods Hospital notes were used to collect data such as biochemistry and ultrasound findings. Patient A 25 year old female with primary infertility undergoing in vitro oocyte retrieval presented to a district general hospital in the UK with severe ovarian hyperstimulation syndrome. During a one week period, the fulmination of the syndrome was rapid with clinical manifestations of enlarged ovaries measuring up to 18cm diameter, 2 litres of ascitic fluid, bilateral pulmonary effusion, renal failure and a 7 fold increase in liver enzymes resulting in an acute hepatic dysfunction. Despite supportive management in an intensive care unit, the patient required transfer to a tertiary liver centre for specialist treatment. Intervention Intravascular volume was restored and thus hepatic dysfunction via supportive measures such as strict fluid resuscitation and Carbergoline, a dopamine agonist. Main outcome measure This report provides further evidence of the complications of ovarian hyperstimulation syndrome. Results 6 weeks post discharge investigations confirmed normal liver function and pathology of the ovaries and pelvis. Conclusions There are few published high quality cases of acute hepatic dysfunction presenting as a complication of severe early onset OHSS. Thus, this report provides further evidence of a potentially fatal complication and can contribute to literature improving our understanding of the pathology and treatment of ovarian hyperstimulation syndrome.