Asian Cardiovasc Thorac Ann 2007;15:249-251
© 2007 Asia Publishing EXchange Ltd
Diaphragmatic Laceration, Partial Liver Herniation and Catamenial Pneumothorax
Antonio Bobbio, MD,
Paolo Carbognani, MD,
Luca Ampollini, MD,
Michele Rusca, MD
U.O. of Thoracic Surgery, Department of Surgical Science, University of Parma, Parma, Italy
For reprint information contact: Antonio Bobbio, MD Tel: 39 34 0687 4733 Fax: 39 05 2199 2019 Email: antonio.bobbio{at}unipr.it, U.O. Chirurgia Toracica, Università di Parma, Azienda Ospedaliera di Parma, Viale Gramsci 14, 43100 Parma, Italy.
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ABSTRACT
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Catamenial pneumothorax is a recurrent pneumothorax temporally associated with menstruation. Pathogenesis remains debated however pleural endometriosis or diaphragmatic abnormalities are almost always present. We report the case of a 35-year-old woman with recurrent right catamenial pneumothorax. At thoracoscopy a large laceration of the diaphragm with partial intrathoracic liver herniation was seen. Treatment involved repair of the diaphragmatic lacerations, and pleurodesis. This report shows that large diaphragmatic defects may be observed in patients with catamenial pneumothorax.
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INTRODUCTION
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Catamenial pneumothorax (CP) is defined as recurrent pneumothorax temporally associated with menstruation. Catamenial pneumothorax has been generally considered a rare entity and the pathogenic mechanisms are a source of controversy.1 Alifano et al2 recently reported that the incidence of CP could be as high as 25% of all spontaneous pneumothoraces in woman of reproductive age referred for surgery. In that study diaphragmatic abnormalities (nodules or small perforations) could be found in all the cases, and histopathologic signs of endometriosis in all but one case.
Herein we present the case of a 35-year-old woman with a recurrent right sided CP. At operation a large diaphragmatic defect with partial intrathoracic liver herniation was identified. This original finding provides further insight into the knowledge of the pathologic abnormalities, and pathogenic mechanisms, of CP.
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CASE REPORT
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A 35-year-old woman was hospitalized for a second recurrence of right pneumothorax two years after a right thoracoscopic pleurodesis. The patient was an active smokerr with a past history of one pack per day for 20 years. Gynecological history noted one pregnancy with normal delivery. She complained of persistent dysmenorrhea with recurrent pelvic pain and associated headache. Previous surgical history included an appendectomy when she was 15 years old, and two laparotomies two years before the current hospitalization. In both instances diffuse peritoneal adhesions were found and no intestinal resection was necessary. The first episode of right pneumothorax had occurred at the age of 32 years and had been treated by pleural drainage. The second episode, on the same side, occurred three months later and urgent percutaneous pleural drainage was also carried out. A thoracic computed tomography scan showed no evidence of intrathoracic abnormalities.
The patient underwent right video-assisted thoracoscopy. Neither subpleural blebs nor pleural abnormality were noted. In this setting an apical pleurodesis with Argon beam coagulation was performed. No details concerning the condition of the diaphragm were reported. One year later a further episode of right pneumothorax occurred and was treated with percutaneous chest tube drainage. Eighteen months later another recurrence occurred and the patient was hospitalized in our department. The last two episodes clearly occurred in the menstrual period, whereas a temporal relationship between menses and the previous episodes could not be determined.
When the patient arrived at our institution, chest X-Ray showed a right-sided apical and basal pneumothorax associated with extensive pleuro-pulmonary adhesions on the axillary line. The right diaphragmatic line presented a superiorly convex round opacity of 4 cm in the major diameter (Figure 1
). A new intervention was planned and the patient underwent video-assisted thoracic surgery with a port for the camera, and a muscle-sparing limited thoracotomy. Several pleuro-pulmonary adhesions were found and freed. Liberation of adhesions on the diaphragmatic aspect of the lower lobe allowed exposure of the diaphragmatic dome. A large laceration, approximately 4 cm in diameter, was observed in the tendinous portion of the muscle with a portion of the liver dome herniated in the pleural cavity (Figure 2). No other abnormalities could be identified, either at a pulmonary or pleural level. The diaphragmatic laceration was repaired with non-absorbable interrupted sutures and a mechanical pleurodesis was performed. The patient’s postoperative course was uneventful and she was discharged on the 5th postoperative day. Postoperative measurements of serum CA125 showed abnormally increased levels (39.2 U·mL–1; normal value lower than 25 U·mL–1).
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Figure 1. Preoperative chest X-Ray: right pneumothorax associated to a right round diaphragmatic opacity.
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Figure 2. Intraoperative view of right diaphragmatic laceration and partial intrathoracic liver herniation.
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DISCUSSION
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To our knowledge, this is the first report in the English literature showing that CP may be associated with a large diaphragmatic laceration and partial liver herniation. Particular focus is centered on radiological chest signs of liver herniation through the diaphragm.
Several mechanisms have been proposed to explain the pathogenesis of CP: spontaneous rupture of blebs, alveolar rupture as a result of prostaglandin-induced bronchiolar constriction, sloughing of endometrial implants of the visceral pleura, transdiaphragmatic passage of air through congenital or (much more frequently) acquired diaphragmatic defects.3 This last mechanism is probably one of the most important ones and according to recently published series accounts for the majority of cases.2–4 Small diaphragmatic defects and nodules represent the most frequently observed alterations in these series. Of note, previous experiences have shown that diaphragmatic fenestration was seen in only 19% to 33% of cases.5
The pathogenesis of these defects is not completely understood. While some patients present with congenital defects, it has recently been shown that histopathologic signs of endometriosis can be identified in the majority of cases. We did not perform a biopsy of the edge of the diaphragmatic defect, so no histologic diagnosis of endometriosis was established. However, we hypothesize that endometriosis was likely based on the clinical history (dysmenorrhea, catamenial pneumothorax, episodes of intestinal obstruction with pelvic adhesions) and the abnormality in serum CA125 levels.
The origin of the pleural endometriosis is possibly related to coelomic metaplasia, lymphatic or vascular embolism of endometrial tissue or transdiaphragmatic migration of endometrial tissue as a consequence of retrograde menstruation and peritoneal circulation.6 However the predominance of right-sided localization and the frequent finding of diaphragmatic abnormalities favors the latter pathogenic mechanism. As recently shown, the implantation of endometrial tissue in the diaphragm can lead to involution of the striated muscular fibers with formation of holes.7 Following this pathogenic cascade, the weakness of the diaphragm could lead to laceration, as inferred and observed in the present report.
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REFERENCES
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- Joseph J, Sahn SA. Thoracic endometriosis syndrome: new observations from an analysis of 110 cases. Am J Med 1996;100:164–70.[Medline]
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- Olive DL, Schwartz LB. Endometriosis. N Engl J Med 1993;328:1759–69.[Free Full Text]
- Alifano M, Cancellieri A, Fornelli A, Trisolini R, Boaron M.. Endometriosis-related pneumothorax: clinicopathologic observations from a newly diagnosed case. J Thorac Cardiovasc Surg 2004;127:1219–21.[Free Full Text]
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ABSTRACT
INTRODUCTION
