The catamenial pneumothorax is defined as the accumulation of air in the pleural cavity that appears in women infrequently and spontaneously with various clinical presentations. Actually, it is considered as an extremely rare entity with few cases described in the literature, that is the reason why the etiology is still discussed. However, a strong association with thoracic endometriosis syndrome has been found. We want to emphasize how the importance of conducting a diagnosis and having a timely management would improve the quality of life of the patient and give a better prognosis of the disease. Thus, a case report of a 38-year-old female patient who was receiving hormone therapy as a treatment for abdominal endometriosis and repetitive pneumothorax was presented. In the video-assisted thoracoscopy we saw diaphragmatic lesions and pneumothorax during the perioperative and postoperative period. Emphasize the importance of a detailed inspection of each intrathoracic organ during the surgical procedure, we also showed how the intraoperative pleurodesis, the placement of a mesh on the diaphragm and the continuity of the hormonal treatment, seems to be an effective therapy to prevent recurrences and have a better control of the disease.
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