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Ann Thorac Surg 2002;74:563-565
© 2002 The Society of Thoracic Surgeons
a Unité de Chirurgie Thoracique, Hôtel-Dieu, AP-HP, Universitè Paris VI, Paris, France
Accepted for publication April 1, 2002.
* Address reprint requests to Dr Alifano, Chirurgie Thoracique, Hôtel-Dieu, 1, Place du Parvis Nôtre-Dame, 75181 Paris Cedex 04, France
e-mail: marcoalifano{at}yahoo.com
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A 25-year-old woman was admitted for dyspnea and right-sided chest pain. She had no tobacco exposure and took no medication. She was nulliparous and had never undergone gynecological procedures. Her past medical history was remarkable for right kidney agenesis without renal function impairment, and two episodes of right-sided spontaneous pneumothorax (6 and 1 months previously) treated by tube thoracostomy. On admission, she was on the 2nd day of her menses.
Chest roentgenogram revealed a partial right-sided pneumothorax. Thoracic computed tomographic scan confirmed the right pneumothorax but did not find any associated anomaly.
Video-assisted thoracoscopy was decided. At exploration, numerous violet implants with tiny holes located in the center of the right diaphragm were found. The lung parenchyma presented no endometriotic lesions but apical unruptured little blebs. The portion of the diaphragm involved was thoracoscopically resected with 45-mm endoGIA stapler (Auto Suture, Norwalk, CT). The diaphragm was lifted anteriorly while stapling across it in order to ensure that the underlying liver was not involved in the staple line. The resected specimen measured 8 x 3 x 1 cm. Apical blebs were also resected by endoGIA stapler and parietal pleural abrasion was performed.
At re-review of the chest x-ray film, we observed two air-filled bubbles, 8 x 5 mm and 1 x 1 mm, respectively, in the middle part of the right diaphragm (Fig 1), which were not visible on computed tomographic scan.
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The postoperative course was uneventful and the patient was discharged on the 7th postoperative day.
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The difficulty in the diagnosis of the condition is also due to lack of suggestive imaging signs. The radiologic sign that we report in the present article probably could be suggestive of the presence of a perforated diaphragm [6]. Such entity is often due to diaphragmatic endometriosis in women and represents the cause of a catamenial pneumothorax in the majority of cases. This radiologic finding seems to argue for the transdiaphragmatic passage of air as the pathogenic mechanism of catamenial pneumothorax. In the absence of a mucus plug in the menstrual period, air is aspirated from the outside, proceeds through the Fallopian tube, and penetrates the chest cavity through the perforating endometrial lesion of the right diaphragm.
Our radiologic sign would probably reflect a phase of the passage of an air bubble from the peritoneum to the chest cavity. Lung basis probably constitutes a transient barrier for such passage, the transient blockage of the bubble on the moment of passage being represented by our radiologic sign. The transient character of the phenomenon probably explains the fact that, in our patient, a computed tomographic scan did not show what was suggested by chest roentgenogram.
Another hypothesis could be the presence of air-filled tiny cavities into the thickness of the diaphragm induced by endometrial implants. Such cavities could rupture during menstruation, provoking the pneumothorax. According to this last hypothesis, our chest x-ray film would show the pneumothorax (caused by one or more ruptured nonvisible bubbles) accompanied by two unperforated bubbles.
In our patient, the right side was involved. There are two explanations for the known right-sided predominance of catamenial pneumothorax [6]: (1) preferential flow of peritoneal fluids (possibly including air) and endometrial tissue from the pelvis along the right paracolic gutter up to the subphrenic space; and (2) the different anatomy of the two upper abdominal quadrants: a large, solid, and relatively fixed liver overlain by the right diaphragm causes a "piston" action, whereas the soft and compressible viscera of the left upper quadrant cannot exert such kind of activity.
In our patient, we did not observe endometrial implants in lung parenchyma and the observed blebs were not ruptured. Furthermore, we did not observe pelvic endometriosis, but it is known that less than one-third of catamenial pneumothorax is associated with pelvic endometriosis; also, previous pelvic surgery or uterus manipulation are surprisingly found in only 11% of cases [1].
This report describes a radiologic sign evoking the catamenial etiology of a pneumothorax. This radiologic sign cannot be misdiagnosed with a colon interposition (Chilaiditi syndrome) which usually shows bigger air-filled stool-containing images, or with a hiatus or diaphragmatic hernia, or with free-air in the case of pneumoperitoneum caused by hollow visceral perforation; all of them have more characteristic radiologic signs in a completely different symptomatic context.
Video-assisted thoracoscopy nowadays represents the surgical approach of choice for spontaneous pneumothorax. As already suggested [5], in the setting of catamenial pneumothorax, it is probably even more useful, allowing complete exploration of the chest cavity, including the diaphragm, which is very difficult to check through a limited axillary thoracotomy. Though simple thoracoscopic pleurodesis seems to provide satisfactory results [1], we think that if endometrial implants are found, their resection should be performed whenever possible, in order to take away endometriosis sources, thus probably limiting further endometrial spreading. In the case of lesions located in the diaphragm, a limited diaphragmatic resection is possible by using endoscopic stapling devices without need of an open approach. Resection of the diseased diaphragm allows its closure, thus treating a pathogenetic mechanism of the catamenial pneumothorax.
Although it has been recognized that surgery provides significantly better results as compared with medical treatment alone [1], the inhibition of sex hormone function by medical treatment is still considered of paramount importance in the treatment of endometriosis [1, 5]. Though it is not known if there is a real need for a medical treatment of isolated thoracic endometriosis after complete resection of lesions, we think that nowadays the association of medical treatment with video-assisted thoracoscopy probably should be considered as the optimal treatment modality for these kinds of patients.
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This article has been cited by other articles:
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  A. Bobbio, R. Trisolini, D. Damotte, and M. Alifano Thoracic endometriosis and catamenial pneumothorax , November 28, 2011; 265 - 281. [Abstract] [Fulltext] [PDF]
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