Introduction and importance: Catamenial pneumothorax (CP) is a condition characterized by recurrent spontaneous pneumothoraxes in women, in temporal pattern with menstrual bleeding. This report presents two cases of CP from a family of three sisters affected by this condition.
Case presentation: The first case involved a 38-year-old woman with a history of recurrent pneumothoraxes. Following the diagnosis of CP, she underwent wedge resection with video-assisted thoracoscopic surgery (VATS). However, due to recurrence and the discovery of a diaphragmatic endometrial nodule, she required nodule excision and diaphragm reconstruction through open thoracotomy. The second case concerned a 47-year-old woman who had a single episode of pneumothorax. After being diagnosed with CP, she also underwent VATS. During the procedure, multiple endometrial diaphragmatic lesions were found, necessitating excision via open thoracotomy. Despite the surgery, the pneumothorax reoccurred, which was managed with a hysterectomy and bilateral salpingo-oophorectomy. At the one-year follow-up, both patients were well.
Clinical discussion: CP is a rare disorder with unclear pathophysiology. Additionally, the impact of genetic factors has not been fully evaluated, and familial cases are rarely reported.
Conclusion: We believe that this report of CP in three sisters suggests a potential genetic role in its occurrence, necessitating further investigation. A better understanding of genetic factors may improve our knowledge of the pathophysiology of CP, ultimately aiding in the management of the condition. We also want to emphasize the importance of collaboration between thoracic surgeons and gynecologists in managing CP, as well as improving prevention of its recurrence.
Keywords: Catamenial pneumothorax; Recurrent pneumothorax; Spontaneous pneumothorax; Thoracic endometriosis syndrome; Thoracotomy; Video-assisted thoracoscopic surgery.
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