Severe ovarian hyperstimulation syndrome (OHSS) leads to changes in laboratory analyte concentrations. Whereas elevated aminotransferase activity is often observed, a cholestatic course with hyperbilirubinaemia and icterus seldom occurs. In this report, the case of a 33 year old patient with polycystic ovary syndrome (PCOS) is described who, after stimulation with human menopausal gonadotrophin (HMG), developed severe OHSS with haemoconcentration, ascites, hydrothorax, elevated aminotransferases, hyperbilirubinaemia and icterus. The patient did not become pregnant and the OHSS regressed, together with the normalization of laboratory and clinical parameters and disappearance of the icterus. During the course of an OHSS cholestasis with icterus may occur, which could be explained by a reactive cholestatic hepatosis as a reaction to the hormonal changes induced by the stimulation therapy.