We report the case of a 51-years-old female who was prophylactically treated with diphenylhydantoin after surgery of an intracranial aneurysm. Twenty-four days after beginning the treatment, a general syndrome appeared, in addition to a diffuse cutaneous exanthema. Four days later, a cholestatic syndrome, compatible with "toxic cholestatic acute hepatitis", was developed. The suppression of the drug, was followed by rapid clinical improvement and gradual normalization of transaminases values, although biochemical cholestasis persisted for months.