Life-Threatening Endometriosis-Related Hemopneumothorax

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CASE REPORT MORCOS ET AL LIFE-THREATENING CATAMENIAL HEMOPNEUMOTHORAX

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Fig 1. Chest computed tomographic scan showing consolidation of right lower lobe surrounding abnormal vessel.

FEATURE ARTICLES

assisted thoracoscopic surgery can be offered to these patients. This technique has an established role in the diagnosis and treatment of intrathoracic lesions. Although challenging in the presence of anomalous anatomy, this procedure is still effective and safe in the hands of experienced thoracic surgeons. There are significant advantages, such as less pain and scarring, shorter hospital stay and, in many cases, early return to work as compared with open surgical procedures.

In conclusion, this case highlights a rare cause of recurrent hemoptysis with diagnostic and treatment challenges. With the availability of minimally invasive surgical techniques, this could be managed without the associated morbidity of conventional thoracotomy.

References 1. Kim TS, Lee KS, Im JG, Goo JM, Park JS, Kim JH. Systemic arterial supply to the normal basal segments of the left lower lobe: radiographic and CT findings in 11 patients. J Thorac Imaging 2002;17(1):34 –9. 2. Tao CW, Chen CH, Yuen KH, Huang MH, Li WY, Perng RP. Anomalous systemic arterial supply to normal basilar segments of the lower lobe of the left lung. Chest 1992;102(5): 1583–5. 3. Stocker JT, Malczak HT. A study of pulmonary ligament arteries. Relationship to intralobar pulmonary sequestration. Chest 1984;86(4):611–5. 4. Yabek SM, Burstein J, Berman W Jr, Dillon T. Aberrant systemic arterial supply to the left lung with congestive heart failure. Chest 1981;80(5):636 –7. 5. Barrett JM, Van Hooydonk JE, Boehm FH. Pregnancy-related rupture of arterial aneurysms [review]. Obstet Gynecol Surv 1982;37(9):557– 66. 6. Ashizawa K, Ishida Y, Matsunaga N, Otsuji H, Sakamoto I, Hayashi K. Anomalous systemic arterial supply to normal basal segments of left lower lobe: characteristic imaging findings. J Comput Assist Tomogr 2001;25(5):764 –9.

Life-Threatening EndometriosisRelated Hemopneumothorax Mohib Morcos, MD, Marco Alifano, MD, Anne Gompel, MD, and Jean-François Regnard, MD Service d’Anatomie et de Cytologie Pathologiques, Unité de Chirurgie Thoracique, and Unité de Gynécologie Médicale, Centre Hospitalier Universitaire Hôtel-Dieu, Paris, France

Catamenial pneumothorax is a relatively rare condition, generally of mild to moderate severity. We report the case Accepted for publication Oct 6, 2005.

Fig 2. (A) Hematoxylin and eosin stained section in the region of dissecting artery. (B) The elastic van Gieson stain clearly demonstrates the split between the elastic media and adventitia. © 2006 by The Society of Thoracic Surgeons Published by Elsevier Inc

Address correspondence to Dr Alifano, Unité de Chirurgie Thoracique, Hôtel-Dieu 1, Place du Parvis Notre Dame, Paris, 75181 Cedex 04 France; e-mail: [email protected].

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of a 29-year-old woman who experienced an episode of life threatening right-sided hemopneumothorax in association with menses. She had already been operated on for recurrent pneumothorax. Treatment of the current episode included urgent tube thoracostomy and iterative thoracotomy, together with lung wedge resection, parietal pleurectomy, and partial diaphragmatic excision. Pathologic examination revealed endometrial implants massively involving the diaphragm, the pleura, and the lung parenchyma. The present report shows that endometriosis-related pneumothorax may be extremely severe. The multiple localizations of endometrial implants in this case may provide a support to the different pathogenic theories of endometriosis-related pneumothorax. (Ann Thorac Surg 2006;82:726 –9) © 2006 by The Society of Thoracic Surgeons

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ecurrent pneumothorax, generally occurring within the menstrual period represents the most frequent manifestation of thoracic endometriosis [1]. Although endometriosis-related pneumothoraxes are frequently known to be mild to moderate, severe presentations in relation to a bilateral disease have been also reported [2, 3]. We hereby present a case of life-threatening endometriosis-related hemopneumothorax. A 29-year-old woman was admitted for right-sided chest pain and mild dyspnea. Her history included two previous right thoracotomies for recurrent pneumothorax. During the second thoracotomy, performed in another institution 8 months prior to the current admission, a partial diaphragmatic resection had been carried out because of the presence of endometrial implants and holes. Ovarian suppression by triptorelin had been initiated postoperatively, but the patient had refused the after monthly injections. With the current admission the patient was on the second day of her menses. She was mildly tachycardic and her oxygen saturation was 92% breathing room air. Chest roentgenogram showed a right-sided loculated pneumothorax without signs of effusion. She suddenly experienced hypotension and worsening of both dyspnea and tachycardia a few hours after admission. Oxygen saturation dropped to 91% with the patient breathing 4 L/min of oxygen. Laboratory investigation revealed a decrease in hemoglobin level (12.8 G/dL, versus 14.7 G/dL on admission) and computed tomographic scan showed a multiloculated hemopneumothorax, with a large loculation being responsible for a mediastinal shift and cardiac compression (Fig 1). Air and serosanguineous fluid (800 mL) were evacuated by emergent tube thoracostomy with a dramatic improvement of symptoms. The patient subsequently underwent thoracotomy. Multiple cysts filled by a serosanguineous fluid were found in the pleural space. Their size ranged from 7 to 12 mm. One of the cysts involved the diaphragmatic dome. A large bulla (2 cm) that was responsible for an air leak was found in the lung apical segment. Several bluebrown lesions ranging from few millimeters to 2 cm were observed on both visceral and parietal pleura. No active

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bleeding could be identified at thoracotomy. A lung wedge resection, an almost complete parietal pleurectomy, and a full thickness diaphragmatic resection of a 5 ⫻ 3 cm area were performed. A 6-month ovarian suppression therapy by triptorelin was introduced. No recurrence of pneumothorax occurred during an 8-month follow-up. Pathologic examination of the diaphragmatic cyst showed a cavity lined with mesothelium, with the presence of endometrial glands embedded within a moderately inflammatory endometrial stroma (Fig 2a). At the periphery of this lesion, endometrial stroma was present on both pleural and peritoneal surfaces of the diaphragm (Fig 2b). In all the cystic lesions of the pleura, endometrial stroma within fibrosis was found (Fig 2c). Lung parenchyma close to the resected apical bulla showed a single endometrial focus (Fig 2d), which was confirmed by immunohistochemistry (Fig 2e), whereas several endometrial foci were found on the surface and the full thickness of the bulla (Fig 2f).

Comment Endometriosis-related pneumothorax was considered as an extremely rare entity until recently. Several reports and some surgical series have been recently published [1, 4], testifying either an increased incidence of the disease or its improved recognition. However, controversies still remain about the exact cause and pathogenesis. It is believed that some cases of catamenial pneumothorax could be secondary to rupture of either bullae or alveoli after a vascular or bronchiolar constriction, or both, caused by paracrine secretion of prostaglandin F2-alpha by endometrial implants [1]. In our patient, several endometrial foci were found in the visceral pleura as well as, more interestingly, in the wall of a large and ruptured lung bulla and in the adjacent lung parenchyma. How endometrial tissue reached the periphery of the lung may be a matter of discussion, but the theory of

Fig 1. Computed tomographic scan showing mediastinal shift and cardiac compression by a large hydro-aeric loculated effusion in the right pleural space.

FEATURE ARTICLES

Ann Thorac Surg 2006;82:726 –9

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CASE REPORT MORCOS ET AL LIFE-THREATENING CATAMENIAL HEMOPNEUMOTHORAX

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FEATURE ARTICLES Fig 2. (a) An endometrial gland and stroma within the diaphragm (Hematoxylin and eosin; ⫻40). (b) Endometrial stroma on peritoneal surface of diaphragm (Hematoxylin and eosin; ⫻20). (c) Endometrial stroma on visceral pleural surface (Hematoxylin and eosin; ⫻20). (d) An endometrial focus within lung parenchyma (Hematoxylin and eosin; ⫻20). (e) Expression of estrogen receptors (left) (immunohistochemistry, primary antibody origin, Clone 6F11, Novocastra, Newcastle, England, ⫻100, oil immersion), and progesterone receptors (right) (immunohistochemistry, primary antibody Clone 16, Novocastra, Newcastle, England, ⫻40). (f) Endometrial subpleural focus within the wall of a bulla (Hematoxylin and eosin; ⫻40).

endometrial vascular embolization [1] may apply to this case. Diaphragmatic endometriosis was also found in our patient. We and some colleagues have recently pointed out that diaphragmatic lesions are an extremely frequent feature of patients with catamenial pneumothorax, and have underlined that the trans-diaphragmatic passage of air is probably the most important mechanism of catamenial pneumothorax [1, 5–7]. Interestingly, in this case we document the presence of endometrial implants on both peritoneal and the pleural surfaces of the diaphragm. It is generally believed that endometrial tissue moves from the pelvis up to the right hemidiaphragm through the right paracolic gutter [1, 5–7]. The pathologic findings in the present case (endometrial tissue on both peritoneal and pleural surfaces, as well as within the muscular layer of the diaphragm), provide a morphologic confirmation to this theory. Therefore this case provides a support to the different pathogenic theories of thoracic endometriosis.

Our patient initially presented with mild symptoms, but experienced a sudden degradation of clinical conditions in relation to an increase in size of the pneumothorax and the hemorrhagic complications. Both of them were in turn responsible for the mediastinal shift. Such presentation of endometriosis-related pneumothorax is exceptional.

References 1. Alifano M, Trisolini R, Cancellieri A, Regnard JF. Thoracic endometriosis: a review of current knowledge. Ann Thorac Surg, in press. 2. Carter EJ, Ettensohn DB. Catamenial pneumothorax. Chest 1990;98:713– 6. 3. Laws HL, Fox LS, Younger B. Bilateral catamenial pneumothorax. Arch Surg 1977;112:627– 8. 4. Korom S, Canyurt H, Missbach A, Schneiter D, Kurrer MO, Haller U, et al. Catamenial pneumothorax revisited: clinical approach and systematic review of the literature. J Thorac Cardiovasc Surg 2004;128:502– 8.

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5. Alifano M, Roth Th, Camilleri Broet S, Schussler O, Magdeleinat P, Regnard JF. Catamenial pneumothorax: a prospective study. Chest 2003;124:1004 – 8. 6. 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. 7. Roth Th Alifano M, Schussler O, Magdeleinat P, Regnard JF. Catamenial pneumothorax: an original chest X-ray sign and thoracoscopic treatment. Ann Thorac Surg 2002;74:563–5.

Tension Chylothorax John B. Ammori, MD, Allan Pickens, MD, Andrew C. Chang, MD, and Mark B. Orringer, MD Department of Surgery, University of Michigan, Ann Arbor, Michigan

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he incidence of chylothorax after a pneumonectomy is 0.5% to 0.7%, and after a transhiatal esophagectomy the incidence is 2% to 4% [1, 2]. Postoperative chylothorax under tension causing respiratory and hemodynamic collapse is exceedingly rare with few such reports in the literature [3, 4]. Two cases of tension chylothorax, one following a pneumonectomy and another following a transhiatal esophagectomy are presented.

Case Reports Patient 1 A 70-year-old man underwent a right pneumonectomy and mediastinal lymph node dissection for a T2N1M0 (stage IIB) bronchogenic squamous cell lung carcinoma at another hospital. He recovered well and was discharged home. Fourteen days postoperatively he presented with drainage from his incision. Thoracentesis revealed a milky effusion characteristic of chylothorax. The patient was transferred our institution. Nonoperative treatment was initially successful. However, on postoperative day 22 he sustained a bradycardic respiratory arrest. A chest roentgenogram demonstrated complete filling of the right hemithorax with a tracheal shift to the Accepted for publication Oct 17, 2005. Address correspondence to Dr Pickens, MD, 2120 Taubman Center, Box 0344, 1500 East Medical Center Dr, Ann Arbor, MI 48109; e-mail: [email protected].

© 2006 by The Society of Thoracic Surgeons Published by Elsevier Inc

Fig 1. A chest roentgenogram demonstrating complete filling of the right hemithorax with a tracheal shift to the left.

left (Fig 1). Right tube thoracostomy evacuated 7 L of chyle under pressure. Chest tube drainage of 3 L to 4 L daily continued for 2 days. A nasoenteric tube was placed, and cream was administered at 90 cc per hour. He was returned to the operating room where the thoracic duct injury was identified, and mass ligation of the thoracic duct was performed at the level of the diaphragm. The chyle leak did not recur. The patient was ultimately discharged to a rehabilitation facility.

Patient 2 A 74-year-old woman underwent a transhiatal esophagectomy and cervical esophagogastric anastomosis for an esophageal perforation complicating an esophagoscopy and dilation of a mid-esophageal stricture. Pathologic analysis revealed T3N0 (stage IIA) squamous cell carcinoma. Eleven days postoperatively she had hemodynamic and respiratory instability. A chest roentgenogram revealed a large right pleural effusion. A right tube thoracostomy evacuated 3.5 L of chyle. The chest tube continued to drain 2 L daily for 2 days. Through a nasoenteric tube, cream was administered at 90 cc per hour. She was returned to the operating room where the thoracic duct injury was identified 5 cm superior to the diaphragm. Mass ligation of the thoracic duct at the level of the diaphragm was performed. The chyle leak did not recur. The patient was discharged home 25 days after her esophagectomy.

Comment Chylothorax complicating a pneumonectomy is typically recognized by an unusually quick filling of the pneumonectomy space in the early postoperative period. Usually 50% to 60% of the pneumonectomy space is filled within 2 weeks of surgery, and 80% to 90% is filled after 6 weeks [3]. Rapid accumulation of fluid under high pressure may lead to hemodynamic and respiratory compromise. Chylothorax after a pneumonectomy is less well tolerated 0003-4975/06/$32.00 doi:10.1016/j.athoracsur.2005.10.033

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Thoracic duct injury resulting in chylothorax is a wellknown and documented complication of thoracic surgery. However, chylothorax under tension is a rarely reported complication that results in respiratory and hemodynamic collapse. Early recognition and treatment of this entity are essential for optimal patient outcome. Herein we present two cases of postoperative tension chylothorax followed by a review of the diagnostic work-up and therapy for this complication. (Ann Thorac Surg 2006;82:729 –30) © 2006 by The Society of Thoracic Surgeons