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Ann Thorac Surg 2003;75:378-381
© 2003 The Society of Thoracic Surgeons

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Original article: general thoracic

Catamenial pneumothorax: retrospective study of surgical treatment

^a Service de Chirurgie Thoracique, Hôpital Européen Georges Pompidou, Paris, France

Accepted for publication August 24, 2002.

* Address reprint requests to Dr Riquet, Service de Chirurgie Thoracique, Hôpital Européen Georges Pompidou, 20-40 rue Leblanc, 75908 Paris, France
e-mail: marc.riquet{at}hop.egp.ap-hp-paris.fr

	Abstract

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BACKGROUND: Catamenial pneumothorax is a rare entity characterized by recurrent accumulation of air in the thoracic space during menstruation. Catamenial pneumothorax is also associated with a high rate of postoperative recurrence. The aim of this study was to discuss the etiology and to determine the optimal surgical treatment of this entity.

METHODS: From December 1991 to September 2000, 10 patients with catamenial pneumothorax were treated at our institution. Median age at time of operation was 37 years (range, 21 to 44 years). We retrospectively evaluated the pathologic findings, the operation performed, and the results in all patients. The mean follow-up was 55.7 months.

RESULTS: Pleurodesis alone was performed in 5 patients and an associated diaphragmatic procedure was performed in 5 patients. In 5 patients, no diaphragmatic anomaly was discovered: 3 experienced one or more recurrences and all still suffer from chronic catamenial chest pain. Hormonal therapy temporarily improved outcome for 6 months in 2 patients. On the contrary, in 5 patients surgical pleurodesis was associated with the repair of diaphragmatic defects (simple closure or coverage by a polyglactin mesh): these patients experienced no recurrence (n = 0/5, p = 0.0016) and no subsequent catamenial chest pain.

CONCLUSIONS: The postoperative outcome is influenced by the diagnosis of diaphragmatic defects with or without endometriosis. Surgical treatment should be accomplished during menstruation for an optimal visualization of pleurodiaphragmatic endometriosis. Because diaphragmatic lesion is frequent and may be occult, we propose the systematic coverage of the diaphragmatic surface by a polyglactin mesh to prevent catamenial pneumothorax recurrence even when the diaphragm appears normal.

	Introduction

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Recurrent spontaneous pneumothorax occurring in association with menstruation was first described by Maurer and colleagues [1] in 1958. Later, Lillington and colleagues [2] named this syndrome catamenial pneumothorax and presented 5 patients in 1972. The reports of catamenial pneumothorax (CPX) in the literature are rare and its physiologic mechanism remains unclear. Traditional therapy involving hormonal treatment or surgical pleurodesis seems to be associated with a high rate of recurrence [3]. The purpose of this retrospective study of 10 patient with CPX is to discuss the pathogenesis and to evaluate different methods of therapy for CPX.

	Material and methods

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From December 1991 to September 2000, 10 patients presenting with CPX underwent surgical treatment for CPX at our institution. The criteria used for diagnosis of CPX were as follows: recurrent pneumothorax documented by chest radiograph during or preceding menstruation and association of recurrent pneumothorax with diaphragmatic endometriosis confirmed histologically. Clinical characteristics are presented in Table 1. Mean age of patients was 37.2 years (range, 21 to 44 years). Pneumothoraces were unilateral and right-sided in 8 patients, left-sided in 1 patient, and bilateral in 1 patient. Symptoms consistent with pelvic endometriosis were observed in 5 of the 10 patients. One patient had a history of catamenial hemoptysis and 5 patients had been previously operated for right recurrent pneumothorax (2 in our institution, 3 at other institutions). We reviewed the different surgical treatment performed and their results using ² analysis. All statistical analysis were performed using computerized software (StatView, Brain Power Inc, Calabasas, CA), with a p value of less than 0.05 considered as significant.

	Results

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	Comment

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Pathogenesis
The mechanism of CPX is unclear. Four proposed mechanisms for the cyclic occurrence of pneumothorax have been described in the literature. The first mechanism involves a congenital diaphragmatic fenestration or porosity. During menstruation, there is an open connection between the atmosphere and the peritoneal cavity because the cervical mucus plug is absent. Air can migrate through the fallopian tube into the abdominal cavity and through diaphragmatic fenestrations or porosities, mainly observed on the right side, causing a pneumothorax [4–6]. The second mechanism involves acquired diaphragmatic fenestrations caused by endometriosis. Endometrial tissue migrates to the diaphragm by retrograde flow from the uterus to the pelvis, hence in the peritoneal cavity to the subphrenic space [7]. When such endometrial tissue undergoes cyclical necrosis due to menstrual hormonal activity, the diaphragm perforates, producing a defect [8]. Endometrial tissue has been identified on the edges of the defect in many cases of CPX [9–11] (and in 5 patients in our population). Pneumoperitoneum can migrate through these defects to cause a pneumothorax during menstruation [12]. The third mechanism involves the metastatic spread of endometrial tissue. Endometrial implants find their way through the uterine veins in the venous system [13]. In this model, endometrial implants reach the lung parenchyma and cause focal defects on the pleural surface during menstruation leading to air leaks [14]. The fourth mechanism cites the release of dinaprost tromethamine (prostaglandin F₂). During menstruation prostaglandin F₂ may be present in the plasma of some women. It is a potent constrictor of bronchioles and vascular structures. Alveolar tissue damaged by vasospasm may cause pneumothorax if bronchospasm impedes expiration [15].

In accord with the above described, our patient population can be divided into two groups.

The first group is characterized by the presence of diaphragmatic defects (5 patients). Diaphragmatic defects were identified during the first procedure in 2 patients and during the second procedure in 3 patients. All of these patients had a history of severe pelvic endometriosis (infertility, chronic pelvic pain). These observations lend support for the mechanism involving diaphragmatic perforation caused by retrograde peritoneal implantation of endometrial tissue. Shiraishi [16] reported that diaphragmatic defects were found in 29% of patients with CPX in the English literature and in 66% of cases in the Japanese literature. In our series, diaphragmatic defects were identified during the second procedure in 60% of the patients (n = 3). Pleurodiaphragmatic endometriosis undergoes cyclical changes due to hormonal activity. During menstruation, implants are congestive, intermittent bleeding causes necrosis, and subsequently the diaphragm perforates. Afterward, the lesions transform into fibrous tissue. Therefore, we recommend a complete exploration of the diaphragmatic surface, with the optimal time for the surgical procedure being the period of menses.

In the second group, diaphragmatic defects were not visualized. One patient with bilateral recurrent CPX had a history of catamenial hemoptysis, which suggested the presence of parenchymal endometriosis. In this patient, lung metastasis of endometrial tissue through the venous system can explain the bilateral localization of parenchymal endometriosis.

Four patients with unilateral recurrent CPX (right-sided in 3 patients and left-sided in 1 patient) had only blebs on the pleural surface. Surgical blebs resection combined with pleurodesis was inadequate as seen by postoperative recurrence in 3 patients and chest pain during menstruation in all patients. In these patients, the mechanism of CPX is still unclear but chest roentgenograms revealed a pneumothorax at the right base in 3 patients, which suggests that the recurrences were probably due to unobserved diaphragmatic lesions.

Treatment
Current treatment of CPX combines the principles of treatment of spontaneous pneumothorax with that of hormonal treatment. Because a direct relation between menstruation and pneumothorax has been demonstrated, prophylactic hormonal therapy used to suppress ectopic endometrium activity by blocking hormonal support from the ovary is warranted. Gonadotropin-releasing hormone agonists induce an hypogonadotropic hypogonadism. The side effects are those of hypoestrogenism, especially osteoporosis, which limits duration of the treatment to 6 months [17]. In our patients, this treatment was not helpful when diaphragmatic repair was not performed. Apical pleurodesis can be achieved using pleural abrasion or pleurectomy, but this method does not prevent diaphragmatic perforation and catamenial chest pain that are, in the majority of the patients, both induced by cyclical proliferation of microscopical endometrial implants [3]. In 3 patients, we lined the diaphragm with a polyglactin mesh. This technique was performed for fear of leaving behind small defects. The goals of polyglactin mesh insertion was to reinforce the diaphragmatic surface, to induce fibrotic adhesion with the lung [18], and to contain and prevent diaphragmatic perforation by endometrial implants. The good results obtained with this technique (no recurrence and no chest pain after the interruption of hormonal treatment for a mean follow-up of 35 months [range, 30 to 45 months]), suggest to use more widely this technique when no diaphragmatic defect is discovered during operation. In fact, recurrence may be due to diaphragmatic defects with or without endometriosis. This was probably the case in our 3 patients who experienced postoperative recurrences at the right base and who may have benefited from this procedure.

To conclude, with respect to our series and reports in the literature, there is considerable evidence to support the involvement of endometriosis in the pathogenesis of CPX. It is important to differentiate between pulmonary endometriosis, which appears to be the result of vascular metastasis, and diaphragmatic endometriosis, which is presumed to be more frequent than reported in the literature. Conventional treatment of CPX appears to be insufficient, but when diaphragmatic lesions are visualized, appropriate treatment significantly improves patient outcome. This observation focuses on the importance of exploration of the diaphragm during menstruation. However, when diaphragmatic endometriosis is suspected during CPX but not observed during operation, we suggest lining of the diaphragm with a polyglactin mesh to help prevent recurrence.

	References

Top Abstract Introduction Material and methods Results Comment References

 

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