A hydrothorax following ventriculoperitoneal shunt catheter insertion is very rare and usually reported in children. Only about 25 cases have been described in the literature and very few have been adults. We report a 51-year-old woman with a massive hydrothorax and respiratory distress following both supradiaphragmatic and transdiaphragmatic migration of a ventriculoperitoneal shunt catheter into the pleural space. To our knowledge this is the first report of the simultaneous occurrence of two types of such migration in one patient.