Asian Cardiovasc Thorac Ann 2005;13:271-273
© 2005 Asia Publishing EXchange Ltd
A Very Rare Entity: Catamenial Pneumothorax
A Serhan Poyraz, MD,
Dalokay Kilic, MD,
Ahmet Hatipoglu, PhD,
Beyhan A Demirhan, PhD1
Department of Thoracic and Cardiovascular Surgery
1 Department of Pathology, Faculty of Medicine, Baskent University, Ankara, Turkey
For reprint information contact: A Serhan Poyraz, MD, Tel: 90 332 257 0606, Fax: 90 332 247 6886. Email: ahmetserhanpoyraz{at}hotmail.com, Department of Thoracic and Cardiovascular Surgery, Faculty of Medicine, Baskent University, Konya, Turkey.
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ABSTRACT
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Catamenial pneumothorax, a variant of spontaneous pneumothorax occurs exclusively in women of menstrual age. Catamenial pneumothorax is associated with a high rate of recurrence. The etiology and pathogenesis is enigmatic. We describe the case of a 42-year-old woman with right-sided catamenial pneumothorax recurring four times, caused by ectopic endometriosis in the pleural layers which was confirmed histopathologically. Surgical treatment should be accomplished during menstruation for optimal visualization of pleurodiaphragmatic endometriosis.
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INTRODUCTION
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Recurrent spontaneous pneumothorax in association with the menstrual cycle was first described by Maurer and colleagues in 1958 and later described as ‘catamenial pneumothorax’ by Lillington and coworkers in 1972.1 ‘Catamenial’ derives from the Greek root meaning monthly. Endometriosis is defined as an extra uterine growth of endometrial tissue and affects about 5% to 10% of fertile women. Thoracic endometriosis is the most common site of the extrapelvic localization. We performed thoracic surgery on a 42-year-old woman presenting with pneumothorax and thoracic endometriosis.
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CASE REPORT
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A 42-year-old woman had 4 episodes of spontaneous pneumothorax. In the first episode, the patient with no significant medical history presented to the casualty department in August 2002 complaining of chest pain of sudden onset and difficulty in breathing. She had no history of fever or hemoptysis. Upon physical examination, she was found to be dyspneic with decreased air entry in the right lung. Laboratory investigations were normal. The patient had no history of thoracic disease, was a non-smoker and in good health. We did not correlate the symptoms with the onset of menses at that time. A chest X-Ray showed 50% pneumothorax without bulla or pleural effusion. She had a chest tube inserted. Four months later, she was admitted and found to have dyspnea again. An X-Ray revealed a recollapse. Right pneumothorax was confirmed and treatment was given as with the first episode.
However, after three months, a third recollapse occurred. Chemical pleurodesis with administration of tetracycline was performed to prevent recurrent basal pneumothorax. After four months, a fourth episode of pneumothorax occurred and we performed a thoracotomy that coincided with the onset of menses. During the operation, adhesion was visualized between the visceral pleura and the chest wall in the right upper lobes of the lung, while air leaks from blebs were visualized in the middle lobe. Holes in the diaphragm and the area with dimples in the visceral pleura were not visualized. We performed blebectomy and partial parietal pleurectomy. The hypertrophic pleura in the apex part were also resected. Ectopic endometriosis in the parietal pleura was confirmed histopathologically (Figure 1
). Postoperative recovery was uneventful, and the patient was discharged on the fourth day after the operation and referred for gynecological review. Gonadotropin releasing hormone analogs (GnRH-analogs) were used for suppressing menstruation and she is currently symptom-free after three months of follow-up.
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Figure 1. Endometrial stroma was seen in the thickened pleura (hematoxylin and eosin stain, magnification x 10).
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DISCUSSION
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Spontaneous recurrent pneumothorax related to menstruation was first described by Maurer and colleagues in 1958. Later, Lillington and coworkers named this syndrome ‘catamenial pneumothorax’ and presented five patients in 1972.1 The etiology and pathogenesis of this syndrome was and still is enigmatic. Four existing theories on pathogenesis include: (1) loss of cervical mucus during the menstrual phase, allowing movement of air into the peritoneum, the air then traverses through the congenital diaphragmatic fenestrations into the pleural space. These acquired diaphragmatic fenestrations are caused by endometriosis. When such endometrial tissue undergoes cyclical necrosis due to menstrual hormonal activity, the diaphragm perforates, producing a defect;2 (2) spontaneous rupture of bullae or blebs during hormonal changes;3 (3) elevated levels of prostaglandin in the serum during menstruation. Prostaglandin-induced bronchiolar constriction results in alveolar rupture; and (4) the metastatic spread of endometrial tissue. Endometrial implants find their way through the uterine veins into the venous system. In this model, endometrial implants reach the lung parenchyma and cause focal defects on the pleural surface during menstruation, leading to air leaks.3
Thoracic endometriosis has been classified as either pleural or parenchymal. Differentiating between pulmonary and pleural endometriosis is important.3 Parenchymal endometriosis usually occurs in women without co-existing pelvic disease and is believed to arise from embolism of endometrial tissue from the uterus to the lungs via the pulmonary arteries. Pleural endometriosis is the more common form, and usually manifests itself as catamenial pneumothorax or hemothorax, the main symptoms being chest pain and dyspnea. Pleural endometriosis is believed to be caused by the regurgitation of endometrial tissue through the fallopian tubes and migration to the pleura through diaphragmatic fenestrations. Most patients with pleural endometriosis also have pelvic endometriosis. In our patient, blebs were found after careful inspection of the lung. However, pathologically confirmed endometriosis in the pleura is extremely rare in the literature.4 Our report supports the theory that catamenial pneumothorax is caused by endometrial implants, which could have traveled across diaphragmatic fenestrations or lymphatic channels to reach the visceral pleura. Pleural endometriosis can be a cause for catamenial pneumothorax.
Catamenial pneumothorax may be suspected based on optimal diagnostic tools and patient symptoms. The age of onset for symptoms is during the mid-thirties. The mean time before diagnosis is 8 months because of delay until attention is brought to the relationship to menstruation. Many women are treated for spontaneous pneumothorax until a correlation is made between multiple lung collapses and the start of menses. Hormonal treatment or pleurodesis is associated with a high rate of recurrence. Thoracotomy is still the gold standard for diagnosis and treatment. Video assisted thoracotomy is not adapted to treatment, a mini thoracotomy is helpful if additional repair of the diaphragm is required. In our case, where blebectomy and partial pleurodesis were performed, video assisted thoracotomy was sufficient as no diaphragmatic lesions were repaired.5
Recurrent catamenial pneumothorax could be due to visceral endometrial implants. Therefore, careful inspection of the entire lung surface at the time of operation for catamenial pneumothorax is mandatory, and thoracotomy should be accomplished during menstruation for an optimal visualization of pleurodiaphragmatic endometriosis. Systemic therapy with GnRH-analogs, pleurodesis, or both should be considered in the treatment of catamenial pneumothorax.
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REFERENCES
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- Lillington GA, Mitchell SP, Wood GA. Catamenial pneumothorax. JAMA 1972;219:1328–32.[Abstract/Free Full Text]
- Kirschner PA. Catamenial pneumothorax: a unifying concept. Ann Thorac Surg 2000;69:1644.[Free Full Text]
- Van Schil PE, Vercauteren SR, Vermeire PA, Nackaerts YH, Van Marck EA. Catamenial pneumothorax caused by thoracic endometriosis. Ann Thorac Surg 1996;62:585–6.[Abstract/Free Full Text]
- Sakamoto K, Ohmori T, Takei H. Catamenial pneumothorax caused by endometriosis in the visceral pleura. Ann Thorac Surg. 2003;76:290–1.[Abstract/Free Full Text]
- Kohiyama R, Maruoka H, Abe N, Ishida H, Yamada S, Ishihara T, et al. Video assisted thoracic surgery for the treatment of catamenial pneumothorax. Kyobu Geka 2000;53:774–9.[Medline]
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