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A case of unilateral congenital hydrothorax diagnosed at 21 weeks and treated with a pleuro-amniotic shunt at 23 weeks' gestation is presented. The rapid production of pleural fluid led to the development of polyhydramnios which was treated with a selective Cox inhibitor. Due to worsening of the hydrothorax, a third thoracocentesis was performed at 30 weeks' gestation prior to a Cesarean section. The neonate responded well to mechanical ventilation, a thoracic drainer that was inserted between days 5 and 14, fluid and protein replacement and gradual transition from parenteral to intestinal nutrition. Early diagnosis of chylothorax should be treated by thoraco-amniotic drainage to prevent fetal pulmonary hypoplasia and congestive cardiac failure. Polyhydramnios is a complication of this therapy and may require treatment with a Cox inhibitor.
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