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To describe a rare case of unilateral massive hydrothorax after controlled ovarian stimulation for IVF and to analyze the diagnostic complexities in a patient lacking both risk factors and typical features of ovarian hyperstimulation syndrome (OHSS). We present a case of a 35-year-old woman suffering from primary infertility due to a severe male factor. Admitted to hospital for dyspnea, the patient initially underwent a thoracentesis. Later, due to the recurrence of massive hydrothorax, permanent pleural drainage was placed. Recognized