Background: Unilateral hydrothorax is rarely the sole manifestation of the ovarian hyperstimulation syndrome (OHSS) and is suggestive of the severity of the disease. Case: A 35-year-old woman presented with mild dyspnea 2 weeks after ovarian stimulation with hMG and hCG and IVF-ET. Chest X-ray revealed a large pleural effusion on the right side. Three consecutive thoracocenteses were needed to drain a total of 6,800 cm3 of fluid. Following drainage, the respiratory symptoms disappeared. An uneventful pregnancy is in progress. Conclusions: Thoracocentesis is safe and efficient for the treatment of hydrothorax and may be repeated as often as necessary. Clinicians should be aware of the possibility of unilateral hydrothorax as the sole symptom of OHSS.

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