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Ann Thorac Surg 1999;68:1413-1414
© 1999 The Society of Thoracic Surgeons

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Case Reports

Visualization of diaphragmatic fenestration associated with catamenial pneumothorax

^a Division of Pulmonary and Critical Care Medicine and Section of General Thoracic Surgery, Mayo Medical Center and Mayo Graduate School of Medicine, Rochester, Minnesota, USA

Address reprint requests to Dr Cowl, Division of Pulmonary and Critical Care Medicine, Mayo Medical Center, 200 First St SW, Rochester, MN 55905
e-mail: cowl.clayton{at}mayo.edu

	Abstract

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Catamenial pneumothorax is a rare entity of unknown etiology characterized by recurrent accumulation of air in the thoracic space during or preceding menstruation. We documented the presence of a diaphragmatic fenestration during thoracoscopy, lending support for hypotheses involving diaphragmatic defects as possible avenues of air collection in the thorax.

	Introduction

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Although cases of spontaneous pneumothorax are relatively common, recurrent pneumothoraces associated with menstruation are rare. Catamenial pneumothoraces, coined from the Greek root meaning monthly [1], were first described in the late 1950s by Maurer and colleagues [2], but its physiologic mechanism remains unclear.

A 39-year-old woman was referred for evaluation of recurrent right-sided pneumothoraces. She was in excellent health 12 years before presentation with no history of pelvic endometriosis before she developed episodes of chest discomfort characterized by a "dull ache" in the right anterosuperior chest. At first, the pain episodes resolved spontaneously. Gradually, however, the pain, dyspnea, and a sensation of a "fluid motion" in her chest persisted. Right-sided apical pneumothoraces were confirmed radiographically several times each year, and, in retrospect, usually at the onset of menstruation. Although frequent, the pneumothoraces were never more than 15% of the hemithoracic volume and the patient had not previously undergone needle or chest tube decompression. Trial therapies with danazol, leuprolide, and medroxyprogesterone acetate were unsuccessful.

Three weeks before presentation the patient noted increasing dyspnea and thoracic pain on exertion, then experienced right-sided neck pressure associated with right lung collapse (Fig 1). Video-assisted thoracoscopic exploration of the right pleural cavity was subsequently performed. A 1-cm circular diaphragmatic fenestration was identified (Fig 2 ) and sutured closed, followed by mechanical pleurodesis. No intrathoracic endometriosis, blebs, or bullae were visualized. Serum prostaglandin levels were normal. Postoperative recovery was uneventful and she remained asymptomatic 9 months after her procedure.

View larger version (151K): [in this window] [in a new window]   Fig 1. Chest radiograph revealed complete right-sided lung collapse in this otherwise healthy 39-year-old woman.

View larger version (91K): [in this window] [in a new window]   Fig 2. Thoracoscopic view of the 1-cm diaphragmatic fenestration at surgical repair.

	Comment

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The mechanism of catamenial pneumothoraces is unclear, but proposed causes have recently been summarized [4, 5]. The anatomic model involves extrusion of the cervical mucous plug at menses resulting in release of peritoneal air that travels through congenital diaphragmatic defects [2]. A metastatic hypothesis differs only in that endometrial implants travel across diaphragmatic fenestrations or lymphatic channels to reach the lung parenchyma and cause focal defects [6, 7]. Physiologic models cite the release of dinaprost tromethamine (prostaglandin F₂), a potent constrictor of bronchioles and vascular structures, as the underlying cause of alveolar rupture causing pneumothorax [8].

Each pathophysiologic model has been discredited because of a lack of a unifying source for each reported patient in the literature. However, multifactorial causes are likely. Direct visualization of a diaphragmatic defect with no evidence of recurrence after pleurodesis in the face of normal prostaglandin levels lends support for proposed causes involving diaphragmatic fenestrations as avenues of thoracic air collection.

	References

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1. Lillington G.A., Mitchell S.P., Wood G.A. Catamenial pneumo-thorax. JAMA 1972;219:1328-1332.[Abstract/Free Full Text]
2. Maurer E.R., Schaal J.A., Mendez F.L., Jr Chronic recurring spontaneous pneumothorax due to endometriosis of the diaphragm. JAMA 1958;168:2013-2014.
3. Schoenfeld A., Ziv E., Zeelel Y., Ovadia J. Catamenial pneumothorax. Obstet Gynecol Surv 1986;41:20-24.[Medline]
4. Carter E.J., Ettensohn D.B. Catamenial pneumothorax. Chest 1990;98:713-716.[Free Full Text]
5. Fonseca P. Catamenial pneumothorax. J Thorac Cardiovasc Surg 1998;116:872-873.[Free Full Text]
6. 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]
7. Kovarik J.L., Toll G.D. Thoracic endometriosis with recurrent spontaneous pneumothorax. JAMA 1966;196:595-597.[Abstract/Free Full Text]
8. Rossi N.P., Goplerud C.P. Recurrent catamenial pneumothorax. Arch Surg 1974;109:173-176.[Abstract/Free Full Text]

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This Article Abstract Full Text (PDF) 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): Claude Deschamps Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Cowl, C. T. Articles by Deschamps, C. Search for Related Content PubMed PubMed Citation Articles by Cowl, C. T. Articles by Deschamps, C.