In case of ovarian hyperstimulation syndrome, the high incidence of dyspnea in relation with ascites and enlarged ovaries should not justify omission of thoracic evaluation. This manuscript reviews the pathogenesis and clinical presentation of hydrothorax following controlled ovarian hyperstimulation. In addition, we describe the case of a 33-year-old woman with a right massive hydrothorax resulting from controlled ovarian hyperstimulation for intracytoplasmic sperm injection.