Ann Thorac Surg 2006;82:726-729
© 2006 The Society of Thoracic Surgeons
Case report
Life-Threatening Endometriosis-Related Hemopneumothorax
Mohib Morcos, MDa,
Marco Alifano, MDb,*,
Anne Gompel, MDc,
Jean-François Regnard, MDa,b,c
a Service d'Anatomie et de Cytologie Pathologiques, Paris, France
b Unité de Chirurgie Thoracique, Paris, France
c Unité de Gynécologie Médicale, Centre Hospitalier Universitaire Hôtel-Dieu, Paris, France
Accepted for publication October 6, 2005.
* Address correspondence to Dr Alifano, Unité de Chirurgie Thoracique, Hôtel-Dieu 1, Place du Parvis Notre Dame, Paris, 75181 Cedex 04 France. (Email: marcoalifano{at}yahoo.com).
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Abstract
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Catamenial pneumothorax is a relatively rare condition, generally of mild to moderate severity. We report the case of a 29-year-old woman who experienced an episode of life threatening right-sided hemopneumothorax in association with menses. She had already been operated on for recurrent pneumothorax. Treatment of the current episode included urgent tube thoracostomy and iterative thoracotomy, together with lung wedge resection, parietal pleurectomy, and partial diaphragmatic excision. Pathologic examination revealed endometrial implants massively involving the diaphragm, the pleura, and the lung parenchyma. The present report shows that endometriosis-related pneumothorax may be extremely severe. The multiple localizations of endometrial implants in this case may provide a support to the different pathogenic theories of endometriosis-related pneumothorax.
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Introduction
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Recurrent pneumothorax, generally occurring within the menstrual period represents the most frequent manifestation of thoracic endometriosis [1]. Although endometriosis-related pneumothoraxes are frequently known to be mild to moderate, severe presentations in relation to a bilateral disease have been also reported [2, 3]. We hereby present a case of life-threatening endometriosis-related hemopneumothorax.
A 29-year-old woman was admitted for right-sided chest pain and mild dyspnea. Her history included two previous right thoracotomies for recurrent pneumothorax. During the second thoracotomy, performed in another institution 8 months prior to the current admission, a partial diaphragmatic resection had been carried out because of the presence of endometrial implants and holes. Ovarian suppression by triptorelin had been initiated postoperatively, but the patient had refused the after monthly injections.
With the current admission the patient was on the second day of her menses. She was mildly tachycardic and her oxygen saturation was 92% breathing room air. Chest roentgenogram showed a right-sided loculated pneumothorax without signs of effusion. She suddenly experienced hypotension and worsening of both dyspnea and tachycardia a few hours after admission. Oxygen saturation dropped to 91% with the patient breathing 4 L/min of oxygen. Laboratory investigation revealed a decrease in hemoglobin level (12.8 G/dL, versus 14.7 G/dL on admission) and computed tomographic scan showed a multiloculated hemopneumothorax, with a large loculation being responsible for a mediastinal shift and cardiac compression (Fig 1). Air and serosanguineous fluid (800 mL) were evacuated by emergent tube thoracostomy with a dramatic improvement of symptoms. The patient subsequently underwent thoracotomy. Multiple cysts filled by a serosanguineous fluid were found in the pleural space. Their size ranged from 7 to 12 mm. One of the cysts involved the diaphragmatic dome. A large bulla (2 cm) that was responsible for an air leak was found in the lung apical segment. Several blue-brown lesions ranging from few millimeters to 2 cm were observed on both visceral and parietal pleura. No active bleeding could be identified at thoracotomy. A lung wedge resection, an almost complete parietal pleurectomy, and a full thickness diaphragmatic resection of a 5 x 3 cm area were performed. A 6-month ovarian suppression therapy by triptorelin was introduced. No recurrence of pneumothorax occurred during an 8-month follow-up.
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Fig 1. Computed tomographic scan showing mediastinal shift and cardiac compression by a large hydro-aeric loculated effusion in the right pleural space.
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Pathologic examination of the diaphragmatic cyst showed a cavity lined with mesothelium, with the presence of endometrial glands embedded within a moderately inflammatory endometrial stroma (Fig 2a). At the periphery of this lesion, endometrial stroma was present on both pleural and peritoneal surfaces of the diaphragm (Fig 2b).
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Fig 2. (a) An endometrial gland and stroma within the diaphragm (Hematoxylin and eosin; x40). (b) Endometrial stroma on peritoneal surface of diaphragm (Hematoxylin and eosin; x20). (c) Endometrial stroma on visceral pleural surface (Hematoxylin and eosin; x20). (d) An endometrial focus within lung parenchyma (Hematoxylin and eosin; x20). (e) Expression of estrogen receptors (left) (immunohistochemistry, primary antibody origin, Clone 6F11, Novocastra, Newcastle, England, x100, oil immersion), and progesterone receptors (right) (immunohistochemistry, primary antibody Clone 16, Novocastra, Newcastle, England, x40). (f) Endometrial subpleural focus within the wall of a bulla (Hematoxylin and eosin; x40).
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In all the cystic lesions of the pleura, endometrial stroma within fibrosis was found (Fig 2c). Lung parenchyma close to the resected apical bulla showed a single endometrial focus (Fig 2d), which was confirmed by immunohistochemistry (Fig 2e), whereas several endometrial foci were found on the surface and the full thickness of the bulla (Fig 2f).
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Comment
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Endometriosis-related pneumothorax was considered as an extremely rare entity until recently. Several reports and some surgical series have been recently published [1, 4], testifying either an increased incidence of the disease or its improved recognition. However, controversies still remain about the exact cause and pathogenesis.
It is believed that some cases of catamenial pneumothorax could be secondary to rupture of either bullae or alveoli after a vascular or bronchiolar constriction, or both, caused by paracrine secretion of prostaglandin F2-alpha by endometrial implants [1]. In our patient, several endometrial foci were found in the visceral pleura as well as, more interestingly, in the wall of a large and ruptured lung bulla and in the adjacent lung parenchyma. How endometrial tissue reached the periphery of the lung may be a matter of discussion, but the theory of endometrial vascular embolization [1] may apply to this case.
Diaphragmatic endometriosis was also found in our patient. We and some colleagues have recently pointed out that diaphragmatic lesions are an extremely frequent feature of patients with catamenial pneumothorax, and have underlined that the trans-diaphragmatic passage of air is probably the most important mechanism of catamenial pneumothorax [1, 5–7]. Interestingly, in this case we document the presence of endometrial implants on both peritoneal and the pleural surfaces of the diaphragm. It is generally believed that endometrial tissue moves from the pelvis up to the right hemidiaphragm through the right paracolic gutter [1, 5–7]. The pathologic findings in the present case (endometrial tissue on both peritoneal and pleural surfaces, as well as within the muscular layer of the diaphragm), provide a morphologic confirmation to this theory. Therefore this case provides a support to the different pathogenic theories of thoracic endometriosis.
Our patient initially presented with mild symptoms, but experienced a sudden degradation of clinical conditions in relation to an increase in size of the pneumothorax and the hemorrhagic complications. Both of them were in turn responsible for the mediastinal shift. Such presentation of endometriosis-related pneumothorax is exceptional.
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References
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- Alifano M, Trisolini R, Cancellieri A, Regnard JF. Thoracic endometriosis: a review of current knowledge. Ann Thorac Surg, in press..
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- Korom S, Canyurt H, Missbach A, Schneiter D, Kurrer MO, Haller U, et al. Catamenial pneumothorax revisitedclinical approach and systematic review of the literature. J Thorac Cardiovasc Surg 2004;128:502-508.[Abstract/Free Full Text]
- Alifano M, Roth Th, Camilleri Broet S, Schussler O, Magdeleinat P, Regnard JF. Catamenial pneumothoraxa prospective study. Chest 2003;124:1004-1008.[Medline]
- Alifano M, Cancellieri A, Fornelli A, Trisolini R, Boaron M. Endometriosis-related pneumothoraxclinicopathologic observations from a newly diagnosed case. J Thorac Cardiovasc Surg 2004;127:1219-1221.[Free Full Text]
- Roth Th, Alifano M, Schussler O, Magdeleinat P, Regnard JF. Catamenial pneumothoraxan original chest X-ray sign and thoracoscopic treatment. Ann Thorac Surg 2002;74:563-565.[Abstract/Free Full Text]
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November 15, 2007;
176(10):
1048 - 1053.
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Abstract
Introduction
