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J Thorac Cardiovasc Surg 2003;126:1204-1207
© 2003 The American Association for Thoracic Surgery
Brief communication |
a Department of Thoracic and Cardiovascular Surgery, Rouen University Hospital-Charles Nicolle, Rouen, France
b Department of Radiology, Rouen University Hospital-Charles Nicolle, Rouen, France
c Department of Anesthesiology, Rouen University Hospital-Charles Nicolle, Rouen, France
Received for publication March 13, 2003; accepted for publication April 24, 2003.
* Address for reprints: Pierre-Yves Litzler, MD, Department of Thoracic and Cardiovascular Surgery, Charles Nicolle University Hospital, 1, rue de Germont, 76000 Rouen, France
pierre-yves.litzler{at}chu-rouen.fr
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A 35-year-old woman with a medical history of hereditary and recurrent epistaxis and lip telangiectasia was admitted with sudden left thoracic pain and dyspnea. There was no recent history of fever or thoracic trauma. Biologic data were as follows: hemoglobin level, 8.8 g/L; hematocrit level, 28%; oxygen saturation, 97%; and fraction of inspired oxygen, 10 L/min. Chest radiography and computed tomographic (CT) scanning revealed a left hemothorax with a suspicion of a vascular malformation in the left lower lobe (Figure 1). There were no aortic or pericardial abnormalities.
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Follow-up angiography at 1 month and CT scanning at 3 months revealed recurrent limited flow in the lingula fistula (Figure 5). Repeat embolization was performed with 2 coils (1 fibered coil of 2 x 50 mm and 1 coil of 2 x 30 mm) placed with a 0.018-inch microcatheter (Boston Scientific). The occlusion was completed without injection of enbucrilate. Follow-up CT scans at 3 months and 2 years showed no recurrence.
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Rupture of pulmonary arteriovenous malformations (PAVMs) is a rare life-threatening complication. Puskas and colleagues1 did not observe any hemothorax in 21 patients treated between 1964 and 1992. Ference and coworkers2 reported only 5 (3.5%) patients with spontaneous hemothorax and 6 with massive hemoptysis in a study of 143 patients with PAVMs and hereditary hemorrhagic telangiectasia.
In most reported cases of PAVMs, the routine approach is preventive transcatheter embolization to avoid complications. These include neurologic sequelae (ie, stroke, transient ischemic attack, cerebral abscess, and seizures) and, less commonly, pulmonary hemorrhage, which could be fatal.3,4
In contrast to reported cases, our patient was treated in a life-threatening situation. The large hemothorax, as well as the previous medical history, prompted us to perform a CT scan to investigate the presence of PAVM. As reported by Martinez and associates,5 spontaneous hemothorax frequently occurs in patients with disease-related coagulopathy or those receiving anticoagulation treatment. Other causes include intrapleural bleeding complicating pneumothorax (ie, disruption of pleural adhesions or torn vascular adhesions), tuberculosis, sarcoidosis, pulmonary infarction, ruptured thoracic aneurysm, and subdiaphragmatic pathology (endometriosis and pancreatic disease). The CT scan carried out on our patient confirmed the presence of PAVMs.
Because the patient's hemodynamic condition was stable, we decided to perform a transcatheter embolization despite the presence of abundant pleural clots and hemothorax.
In our case the arteries entering the PAVMs were technically suitable for coil occlusion, being long enough and not enlarged just before entry into the aneurysms, without significant risk of the coils entering the aneurysms and the draining veins. When feeding arteries are short (<3 cm), occlusion of the aneurysm might be necessary.6 Detachable coils can be withdrawn if they are not correctly sized or can be reinserted if not properly placed. However, some authors prefer to use detachable balloons.3
Supplying arteries with a diameter of 5 to 9 mm (approximately 70% of PAVMs) can be occluded equally well with either detachable silicone balloons or coils. Personal experience and preference for a certain device will often be decisive in the choice of technique.7 Because anatomy can vary greatly, both detachable silicone balloons and coils should be available as therapeutic options.4 After deployment of several coils in the feeding artery, flow is reduced, and embolization can safely be achieved with glue, which can shorten the procedure.
In most cases PAVMs are multiple, and more than one embolotherapy session is required per patient. In a study reported by White,6 276 PAVMs were diagnosed in 76 patients; all patients had multiple malformations, and on average, 2 embolotherapy sessions were necessary per patient in addition to diagnostic angiography, which was performed on a separate day. Embolotherapy sessions were separated by 4 to 6 weeks.
Current indications for transcatheter embolization include the prevention of complications in larger (feeding artery diameter >3 mm) and symptomatic PAVMs. From our experience in cases of massive hemoptysis-hemothorax, we believe occlusion of all existing PAVMs should be considered. This approach might have the additional benefit of reducing the risk of paradoxical emboli and other complications associated with unoccluded PAVMs.2 To our knowledge, no data are currently available to predict which PAVMs is most likely to rupture on the basis of size or location.
After embolization, different therapeutic options were available. A thoracic drainage procedure without surgical intervention was an option; however, drainage efficiency could be hindered by the presence of numerous clots. A surgical procedure to remove clots and confirm the absence of bleeding was therefore considered essential. Two surgical strategies were available. The first strategy was a thoracotomy with local excision of all the PAVMs. This technique, described by Bosher and colleagues,8 can be combined with a previous embolization procedure.9 The feasibility of selective surgical resection depends on the PAVM localization, however, and lung tissue sparing is not always achievable. The second option was to perform video-assisted surgery with wedge resection of the PAVM, which could avoid the higher morbidity rates of thoracotomy. This technique has been previously described by Temes and associates10 but only for a solitary malformation. In our case the PAVMs were multiple and not easily accessible for video-assisted thoracoscopic resection. Because of a complete absence of PAVM bleeding, we decided only to remove blood clots.
As previously described by several authors,11,12 we observed a recanalization of a PAVM 3 months later. Usually, occluded PAVMs disappear or are reduced to a fibrous strand by the end of 1 year. Any evidence of persistence on CT suggests recanalization (16% in the study of Ference and coworkers2) and is an indication for re-embolization.3 Thereafter, follow-up thoracic CT scanning is recommended every 3 to 5 years to investigate the possible development of new PAVMs or growth of small PAVMs.3 Some authors have proposed the use of 2-dimensional contrast echocardiography, with good results in detection and follow-up.13,14
All patients with hereditary hemorrhagic telangiectasia should undergo routine screening for PAVM with noninvasive techniques, preferably with the 100% oxygen method. Family members of patients with hereditary hemorrhagic telangiectasia should also be screened for PAVM.15
In our case a medical history of hereditary and recurrent epistaxis, as well as the presence of lip telangiectasia, was sufficient to suspect ROW syndrome and screen for PAVMs, which are associated with ROW in 60% to 90% of cases.16
Conclusion
Even in the presence of PAVM rupture, transcatheter embolization could be performed. This procedure, combined with video-assisted thoracoscopy, avoided lung tissue loss, preserved lung function, and minimized morbidity. Because PAVMs can be recurrent, minimally invasive treatment options are essential. Although repeated procedures might be required for persistent PAVMs or recanalization, transcatheter embolization is a lung-sparing procedure and should therefore always be considered in cases of PAVM.
Acknowledgments
We thank Mr Richard Medeiros and Miles Dalby for their advice in editing the manuscript.
References
This article has been cited by other articles:
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  J. Nakajima, S. Takamoto, E. Takeuchi, T. Fukami, and A. Sano Thoracoscopic Surgery for Pulmonary Arteriovenous Malformation Asian Cardiovasc Thorac Ann, October 1, 2006; 14(5): 412 - 415. [Abstract] [Full Text] [PDF]
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