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© 2004 European Association of Cardio-Thoracic Surgery
Huge arteriovenous malformation due to chronic infection o lub
a Department of Thoracic Surgery, Gaziantep University Medical School, 27070 Kolejtepe, Gaziantep, Turkey
* Corresponding author. Tel.: +90-342-335-7273; fax: +90-342-336-5505 Received August 19, 2003; received in revised form November 22, 2003; accepted November 28, 2003
Acquired pulmonary arteriovenous malformations (AVMs) related to chronic infections are extremely rare. We report a 32-year-old male patient having a huge pulmonary AVMs being fed with multi-systemic vascular divisions weeded in ascending aorta due to chronic infection. The AVMs were managed by combined intra-arterial embolization and surgical resection.
Key Words: Pulmonary arteriovenous malformation; Chronic infection; Intra-arterial embolization
Pulmonary arteriovenous malformations (AVMs) were first described by Churton in 1897. Acquired AVMs caused by chronic infections are quite rare (tuberculosis, actinomycosis, schistosomiasis). In this paper, we report the combined surgical resection of a case with arterial embolization that had developed on the ground of a chronic infection.
Thirty-two-year-old male patient was taken under observation with the complaints of lost weight, chest pain, cough and hemoptysis. He continued to have frequent epileptic seizures. He did not report any previous thoracic trauma or infection. He did not have any history of smoking or any exposure to asbestosis. He experienced pain on the left side of his chest 2 months prior to his admission to our hospital. He then developed a cough, which aggravated in the following days. One month before his admission, he had hemoptysis in the form of minimally fresh blood. The patient did not report any difficulty in breathing. His physical examination did not yield any pathological finding. In the laboratory analysis, other than white blood cell (WBC) of 12 000 mm–3 and erytrocide sedimentation rate of 54 mm/h, the results were normal. Chest films demonstrated homogeneous increase of density in the upper, middle and lower zones of the left lung. Echocardiography did not demonstrate any pathological finding other than the moderate enlargement of left chambers. Tuberculin skin test was negative. In the Gram staining and M. tuberculosis examination of the sputum samples, significant findings could not be obtained. Thoracic computed tomography examination yielded a mass lesion on the left lung that started from the posterior part of the upper lobe extending to the diaphragm through paravertebral sulcus while having parenchymal invasion especially on the lower lobe. The lesion did not invade the thoracic wall or have a relationship with the spinal canal. There were not any suspicious mediastinal lymph nodes (Fig. 1).
Bronchoscopic examination did not reveal any pathological finding. Bronchoalveolar lavage and sputum cytology specimens obtained from the left lung did not demonstrate any malignancy. Biopsies through thoracic route revealed findings of chronic infection. We could not reach a conclusion with the examinations performed, thus with the decision of the hospital council we decided to have an open biopsy with mini-thoracotomy. During the open biopsy procedure there was bleeding of high-flow rate from the mass. Hemodynamics could only be stabilized with the transfusion of three units of blood. We arrived at the diagnosis of ‘Bronchiolitis obliterans organizing pneumonia’ with the microscopic examination of the biopsy sample. Aortography showed that the mass was being fed by the six major intercostal branches originating from the aorta; each branch had a collateral vascular network extending to the parenchyma and the thoracic wall. There was also venous drainage to the inferior pulmonary vein with a separate branch from parenchyma. The mass lesion was diagnosed as a systemic-to-pulmonary giant pulmonary AVM with wide collateral network. Intra-arterial embolization was performed together with aortography before the surgical resection (Fig. 2).
The patient underwent angiography with a right common femoral approach under local anesthesia. Initial thoracic aortography showed tortuous and dilated intercostal vessels feeding a giant hypervascular mass lesion in left hemithorax. Next, a 5-French vertebral catheter (BARNH; Boston Scientific, Watertown, MA) was inserted into the each feeding artery and contour polyvinyl alcohol particles (Target Therapeutics Fremont, CA) measuring 355–500 and 500–710 µm mixed with saline and contrast medium (Iomeron 400 [iomeron]; Bracco, Milano, Italy) were infused slowly under fluoroscopy. Each feeding artery was embolized using the same procedure. The patient was taken into the operation for the purpose of resection 48 h after the embolization. Thorax was entered through median sternotomy. The mass lesion was observed to invade the hilar portion of the left lung. Left main pulmonary artery, left superior and inferior pulmonary veins were ligated intra-pericardially. There was an extra-anatomical branch draining to the inferior pulmonary vein from parenchyma. Left main bronchus was divided and closed. Descending aorta was approached, six arterial branches feeding the mass that originated from descending aorta at the level between the third and seventh ribs were ligated and divided. The mass was separated from the adhesions on the diaphragm and the thoracic wall. In the histopathological evaluation, there were findings of chronic infection and the tissue sample correlated with arteriovenous malformation. There was no bacterial growth in tissue cultures. The patient did not develop any complications during the post-operative period. The patient has been asymptomatic during the last 1 year and his epileptic seizures have decreased significantly compared to the pre-operative period.
AVMs can be classified as congenital and acquired depending on the etiological causes [1]. Acquired cases can be observed following trauma, long-standing hepatic cirrhosis and metastatic carcinoma [2–4]. Acquired pulmonary AVMs related to chronic infection (tuberculosis, actinomycosis, schistosomiasis) is very rare [1]. Although there was no growth in tissue cultures, the giant pulmonary AVMs case with widespread parenchymal invasion that we present was shown to have developed on the grounds of a chronic infection together with the histopathological examination findings. We think that pulmonary AVMs has developed on the basis of neovascularization caused by chronic infection. The complaint of dyspnea is reported at a frequency of approximately 50% in the series by Swanson et al., yet it was not present in our patient [5]. In his history, the patient had epileptic seizures refractory to medical treatment. Transient ischemic attacks, hemiplegia, epileptic episodes and brain abscesses are the complications that are frequently observed in pulmonary AVMs cases [6]. Following the operation, the epileptic seizures have decreased significantly. Systemic feeding of pulmonary AVM is a rare condition. It can be fed directly from the aorta in the systemic relationship as well as from internal mammarian artery, intercostal and bronchial arteries [7]. In our case, systemic feeding was via intercostal arteries from aorta, while for the venous return in addition to the intra-parenchymal pulmonary route, it was opening to the left atrium via inferior pulmonary vein through an extra-anatomical branch. For the treatment of pulmonary AVMs, surgical resection, arterial ligation, selective embolization and combined treatment approaches have been tried. Detachable silicon balloons, coils and various medical agents were used for the purpose of embolization [7]. Widespread collateral circulation of significant degree on the chest wall creates an important obstacle to surgery [1,7]. In our case, we had a mass lesion with extensive collateral circulation located on the thoracic wall having significant parenchymal involvement. For the purpose of providing a controllable bleeding environment during resection, embolization was performed. In conclusion, the neovascularization of the surrounding tissue as a result of chronic infection caused a systemic-to-pulmonary AVMs with extensive collateral circulation network that was diagnosed with radiological examination. It was treated with a combination of embolization and surgery. For the treatment of AVM cases with extensive collateral circulation on the thoracic wall and the parenchyma, we believe that combined treatment is a safe approach for achieving complete resection.
ICVTS on-line discussion Author: Dr. Erkan Yildirim, Ankara Numune Education and Research Hospital, Division of Thoracic Surgery, Talatpasa Bulv./Samanpazari, Ankara, Turkey Date: 31-Dec-2003 Message: I would kindly ask the authors to comment on two topics. First, would only blood transfusion be enough to control the bleeding at the biopsy site? Or, did they perform any additional intervention to support controlling bleeding locally? Second, I would like to know when the aortography and embolization was performed? It is not clear in the manuscript. doi:10.1016/j.icvts.2003.11.012
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Abstract
1. Introduction
