Isolated pleural effusion is a rare condition in a fetus or neonate with high mortality. When there are no other findings of hydrops fetalis or documented etiology such as inflammatory, iatrogenic or cardiac problems exist, isolated pleural effusion is considered. Timely diagnosis and management not only avoids mortality but also results in excellent prognosis. For fetal hydrothorax, intrauterine management is usually recommended. For those who present late, postnatal management includes intubation, thoracocentesis, ventilation and supportive care. The authors present isolated bilateral severe hydrothorax in a preterm neonate that resulted in severe respiratory compromise at birth. A single postnatal thoracocentesis resulted in complete resolution. No definite etiology for hydrothorax could be established. He had normal growth and development during his follow up till 1 year of age.
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