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Ann Thorac Surg 2003;76:290-291
© 2003 The Society of Thoracic Surgeons

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Case report

Catamenial pneumothorax caused by endometriosis in the visceral pleura

^a Department of Respiratory Surgery, Yokohama Rosai Hospital, Yokohama, Japan

Accepted for publication December 31, 2002.

^* Address reprint requests to Dr Sakamoto, First Department of Surgery, Yokohama City University School of Medicine, 3-9 Fukuura, Kanazawa-ku, Yokohama 236-0004, Japan
e-mail: saka784{at}lycos.jp

	Abstract

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Catamenial pneumothorax is a rare clinical entity of unknown etiology. The most well known hypothesis is passage of air from the genital tract through endometrial fenestrations in the diaphragm. Although some reports are associated with diaphragmatic endometriosis, few have been confirmed endometrial implants in the visceral pleura. We describe a very rare case of catamenial pneumothorax caused by ectopic endometriosis in the visceral pleura confirmed histopathologically in a woman 1-year after hysterectomy.

	Introduction

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Catamenial pneumothorax (CP) is a rare disease, and its pathogenesis is poorly understood. Various theories have been formulated to explain the development of CP. One of the most widely known theories is that air passes through the genital tract into the thorax through the peritoneal cavity and diaphragmatic fenestrations [1–4]. We report a case of CP that developed after hysterectomy.

A 41-year-old woman had had a hysterectomy for a uterine leiomyoma and monthly abdominal discomfort associated with her menses. Abdominal endometriosis was confirmed at the operation. Postoperatively, the patient still had monthly abdominal discomfort; however, no medication for endometriosis was prescribed. One year after hysterectomy, monthly episodes of chest pain with abdominal discomfort developed. On evaluation of the chest pain, she was found to have a small right-sided pneumothorax. She had three documented episodes of right-sided pneumothorax, occurring at about 2-month intervals during a 5-month period. These pneumothoraces were small and treated conservatively. On the third episode of pneumothorax we performed thoracoscopic surgery.

A small area of fine adhesion was visualized between the visceral pleura and the chest wall in the right upper lobe of the lung. Several blue-brown spots, less than 3-mm diameter, were found on the tendinous portion of the diaphragm (Fig 1). There were also several dark red dimples, less than 5-mm diameter, on the visceral pleura of the lateral aspect of the upper lobe. No blebs or bullae were visualized. The adhered area of the lung, one of the areas with a small dimple in the upper lobe, and the lesions in the diaphragm were resected with endo-staplers. We widely covered the staple line of the lung and the diaphragm with absorbable mesh sheets. Pathologically, ectopic endometrial tissue was found in the resected lesions on the diaphragm. The portions of the lung resected from the area with adhesion and the area with dimples also contained endometrial tissue in the visceral pleura (Fig 2). Postoperative recovery was uneventful, and the patient was discharged on the third postoperative day.

View larger version (163K): [in this window] [in a new window]   Fig 1. Thoracoscopic view of several small blue-brown spots (arrows) on the tendinous part of the diaphragm.

View larger version (143K): [in this window] [in a new window]   Fig 2. Microphotograph illustrating endometrial tissue in the thickened visceral pleura (hematoxylin and eosin stain, magnification x40).

	Comment

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Four theories for the pathogenesis of CP have been proposed: (1) loss of cervical mucus during the menstrual phase, allowing movement of air into the peritoneum, then the air traverses through the diaphragmatic fenestrations into the pleural space [1–4]; (2) spontaneous rupture of bullae or blebs during hormonal changes [2]; (3) elevated levels of prostaglandin in the serum during menstruation, which could cause severe vascular and bronchoalveolar constriction, producing alveolar rupture [2, 4]; and (4) ectopic endometrial tissue in the visceral pleura and its desquamation during menstruation [4, 5].

Given that our patient had previously had a hysterectomy, it is unlikely that the CP was caused by movement of air through the cervix. In our patient, no blebs or bullae were found after careful thoracoscopic inspection of the lung. Thus, our report supports the theory that CP is caused by endometrial implants, which could have traveled across diaphragmatic fenestrations or lymphatic channels to reach the visceral pleura [2, 3]. A common theory regarding the cause of endometriosis is that viable endometrial cells can reflux through the fallopian tubes during menstruation, implant, and grow on the surrounding pelvic structures and peritoneum [6]. These cells could implant in the visceral pleura through the fenestrations in the diaphragm; however, pathologically confirmed endometriosis in the visceral pleura is extremely rare in the literature [5]. Our case confirms that visceral pleural endometriosis can be a cause for CP. Previous case reports of recurrent CP after diaphragmatic resection could theoretically be due to visceral endometrial implants. Therefore, careful inspection of the entire lung surface at the time of operation for CP is mandatory and systemic therapy with GnRH-analogs [2, 4], pleurodesis, or both should be considered on the treatment for CP.

	Acknowledgments

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We thank Shunji Kawamura, MD, for pathologic support.

	References

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1. Lillington G.A., Mitchell S.P., Wood G.A. Catamenial pneumothorax. JAMA 1972;219:1328-1332.[Abstract/Free Full Text]
2. Van Schil P.E., Vercauteren S.R., Vermeire P.A., Nackaerts Y.H., Van Marck E.A. Catamenial pneumothorax caused by thoracic endometriosis. Ann Thorac Surg 1996;62:585-586.[Abstract/Free Full Text]
3. Cowl C.T., Dunn W.F., Deschamps C. Visualization of diaphragmatic fenestration associated with catamenial pneumothorax. Ann Thorac Surg 1999;68:1413-1414.[Abstract/Free Full Text]
4. Blanco S., Hernando F., Gómez A., González M.J., Torres A.J., Balibrea J.L. Catamenial pneumothorax caused by diaphragmatic endometriosis. J Thorac Cardiovasc Surg 1998;116:179-180.[Free Full Text]
5. Flieder D.B., Moran C.A., Travis W.D., Koss M.N., Mark E.J. Pleuro-pulmonary endometriosis and pulmonary ectopic deciduosis: a clinicopathologic and immunohistochemical study of 10 cases with emphasis on diagnostic pitfalls. Hum Pathol 1998;29:1495-1503.[Medline]
6. Rock J.A., Markham S.M. Pathogenesis of endometriosis. Lancet 1992;340:1264-1267.[Medline]

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