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Ann Thorac Surg 1996;62:585-586
© 1996 The Society of Thoracic Surgeons

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Case Report

Catamenial Pneumothorax Caused by Thoracic Endometriosis

Division of Thoracic and Vascular Surgery, Departments of Surgery, Pulmonary Medicine, Radiology, and Pathology, University Hospital of Antwerp, Edegem, Belgium

Accepted for publication March 6, 1996.

	Abstract

Top Footnotes Abstract Introduction Comment Acknowledgments References

 
A 28-year-old woman presented with monthly returning thoracic pain and cough. Catamenial pneumothorax was diagnosed. Thoracoscopy showed multiple nodules on the diaphragm, parietal pleura and lung itself, which proved to be thoracic endometriosis. Thoracoscopy is a useful procedure for diagnosis and treatment of this rare condition.

	Introduction

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A recurrent spontaneous pneumothorax concurrent with the onset of menstruation was first described by Maurer and associates [1] in 1958. Lillington and colleagues [2] introduced the term "catamenial pneumothorax" in 1972. This syndrome is characterized by the appearance of a pneumothorax within 48 to 72 hours after the onset of menses. There is a right-sided predominance of more than 90% [3–5]. The definite cause has not been established, and various theories exist.

A 28-year-old woman presented with a monthly returning cough and right thoracic pain irradiating to the right arm, throat, and back. Except for recurrent genital herpes infections her history was negative. She was nulliparous with a normal menstrual pattern and did not smoke or use any medication. The monthly returning complaints were closely related to the menses. A right apical pneumothorax was documented on four occasions. Computed tomography of the thorax showed no underlying abnormalities of the lung parenchyma. A gynecologic examination revealed no signs of endometriosis externa. Oral contraceptives for 3 months followed by a progestagen for another 3 months did not bring any improvement. Normal levels of ₁-antitrypsin were found.

For refinement of diagnosis a thoracoscopic procedure was performed. The camera was inserted in the midaxillary line in the fifth intercostal space, with two additional ports in the anterior axillary line and behind the scapula. There were no pleural adhesions. On the dorsal parietal pleura, the tendinous part of the diaphragm, and the pulmonary parenchyma itself, many tiny blue-brown nodules were present, which we suspected to be endometrial foci. Several biopsy specimens of these "gunshot" lesions were taken, and a nodular zone of the lower lobe was stapled with the Endo-GIA 60 stapler (United States Surgical Corp, Auto Suture International, Norwalk, CT). On the apex of the right upper lobe fibrotic scar tissue representing old ruptured blebs was noted, and this area was also stapled. In the apical zone a parietal pleurectomy was performed, followed by a pleural abrasion from the third to the sixth rib. No air leakage was present, and the lung fully expanded.

Postoperative recovery was uneventful, and therapy with a gonadotropin-releasing hormone analogue was started. Pathologic examination showed fibrosclerotic tissue, macrophages with iron pigment, and glands of the endometrial type surrounded by endometrial stroma on the parietal pleura, on the diaphragm, and in the lung biopsy specimen of the lower lobe (Fig 1). In the apical segment of the upper lobe interstitial fibrosis was noted together with emphysematous changes of the alveoli.

View larger version (77K): [in this window] [in a new window]   Fig 1. . Microphotograph showing endometrial glands (arrow) in a biopsy specimen of the parietal pleura. (Hematoxylin and eosin; x60 before 35% reduction.)

	Comment

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During menstruation there exists an open connection between the outside and the peritoneal cavity as the cervical mucus plug is absent. Air can then make its way through the tubae, abdominal cavity, and congenital fenestrations in the diaphragm and cause a pneumothorax. Evidence to support the greater frequency of right-sided congenital defects in the diaphragm can be found in Meig's syndrome, where the pleural effusion is always right-sided [3]. Pneumothorax and hemothorax secondary to pneumoperitoneum and hemoperitoneum have only been reported on the right side [3].

Second, high serum levels of prostaglandin F₂ in menstruating women may cause vasospasm and bronchospasm with possible rupture of alveoli, which result in a pneumothorax. The third explanation implies that blebs or bullae, which are usually found in primary spontaneous pneumothorax, would be more susceptible to rupture through hormonal changes. Finally, there is the possibility of thoracic endometriosis. Four theories try to explain the existence of extrauterine endometrial foci [1, 5, 6–8]. According to the implantation theory, menstrual blood with endometrial fragments could regurgitate in the pelvis. This blood could find its way to the right subphrenic space via the right paracolic region and through the above-described fenestrations in the diaphragm [6]. Another pathway could be a transdiaphragmatic lymphatic channel [6]. During uterine manipulation, as with cesarian section or curettage, endometrial fragments could penetrate the lymphatic and venous system. Finally, the celomic metaplasia theory states that endometrium, peritoneum, and pleura are formed out of the primitive peritoneum, and metaplasia could lead to clinical overt endometriosis externa.

Pleural and diaphragmatic endometriosis gives rise to pneumothorax, pleural effusion, hemothorax, and pleural pain [6, 7]. Parenchymal endometriosis may be responsible for hemoptysis. A rare case of catamenial pneumomediastinum has also been described [7]. The percentage of associated pelvic endometriosis varies from 20% to 70% [6].

The possible mechanisms by which thoracic endometriosis produces an air leak every month remain elusive. Diaphragmatic endometrial implants can give rise to holes in the diaphragm through which air passes from the genital tract into the thorax during menstruation. Desquamation of endometrial tissue on the visceral pleura is another possible mechanism causing small pulmonary air leaks resulting in pneumothorax [3, 6].

Treatment of catamenial pneumothorax starts with an oral contraceptive, a progestative, or a gonadotropin-releasing hormone analogue like busereline for 6 months [5, 6, 8]. When this is unsuccessful, a thoracoscopic procedure is the next logical step: biopsy specimens of nodules suspected of being endometriosis after thorough exploration can be taken and a parietal pleurectomy or pleural abrasion performed to obtain pleurodesis. Blebs or bullae, if present, can be stapled or a ligature placed at the base. In this way thoracoscopy is a useful procedure for definite diagnosis and treatment of catamenial pneumothorax.

	Acknowledgments

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We thank Ms Renelde Ruelle for typing the manuscript.

	Footnotes

Top Footnotes Abstract Introduction Comment Acknowledgments References

 
Address reprint requests to Dr Van Schil, Division of Thoracic and Vascular Surgery, Department of Surgery, University Hospital of Antwerp, Wilrijkstraat 10, B- 2650 Edegem, Belgium.

	References

Top Footnotes Abstract Introduction Comment Acknowledgments References

 

1. Maurer ER, Schaal JA, Mendez FL. Chronic recurring spontaneous pneumothorax due to endometriosis of the diaphragm. JAMA 1958;168:2013–4.
2. Lillington GA, Mitchell SP, Wood GA. Catamenial pneumothorax. JAMA 1972;219:1328–32.[Abstract/Free Full Text]
3. Slasky BS, Siewers RD, Lecky JW, Zajko A, Burkholder JA. Catamenial pneumothorax: the roles of diaphragmatic defects and endometriosis. AJR 1982;138:639–43.[Abstract/Free Full Text]
4. Carter EJ, Ettensohn DB. Catamenial pneumothorax. Chest 1990;98:713–6.[Free Full Text]
5. Dattola RK, Toffle RC, Lewis MJ. Catamenial pneumothorax: a case report. J Reprod Med 1990;35:734–6.[Medline]
6. Shiraishi T. Catamenial pneumothorax: report of a case and review of the Japanese and non-Japanese literature. Thorac Cardiovasc Surg 1991;39:304–7.[Medline]
7. Laraqui CH, You B, Mayeux D, Benhamou M, Paitel JP, Grilliat JP. Hémoptysie cataméniale par endométriose broncho-pulmonaire. Rev Mal Resp 1989;6:377–80.
8. Slabbynck H, Laureys M, Impens N, De Vroey P, Schandevyl W. Recurring catamenial pneumothorax treated with Gn-RH analogue. Chest 1991;100:851.[Abstract/Free Full Text]

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This Article Abstract Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Paul E. Van Schil Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Schil, P. E. V. Articles by Van Marck, E. A. Search for Related Content PubMed PubMed Citation Articles by Schil, P. E. V. Articles by Van Marck, E. A.