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J Thorac Cardiovasc Surg 2006;132:707-708
© 2006 The American Association for Thoracic Surgery
Brief Communication |
Division of General Thoracic Surgery, Fujita Health University School of Medicine, Toyoake, Aichi, Japan.
Received for publication January 18, 2006; accepted for publication February 13, 2006. * Address for reprints: Takashi Suda, MD, Division of Thoracic Surgery, Fujita Health University School of Medicine, 1-98 Dengakugakubo Kutsukake Toyoake, Aichi, Japan 470-1192 (Email: suda{at}fujita-hu.ac.jp).
Pulmonary sequestration can present various clinical manifestations, and some patients even lack aberrant arteries. Video-assisted thoracoscopic surgery (VATS) is a useful surgical technique for treating pulmonary sequestration if the vascular supply of the lesion can be accurately characterized. We report here a rare case of extralobar pulmonary sequestration without aberrant arteries, which was treated by VATS.
Clinical Summary
The patient was an 11-year-old boy whose chief symptoms were cough, fever, and pain in the left side of the chest when he visited our hospital. A chest x-ray film revealed a left pulmonary cyst and fluid accumulation in the cyst. An elevated inflammatory response was observed, including a leukocyte count of 13,000/µL and C-reactive protein concentration of 0.9 mg/dL. Computed tomographic (CT) scans revealed a sequestered lung segment in the left upper lobe, in which a pulmonary cyst was observed. Fluid accumulation was also observed in the cyst (Figure 1). Administration of an antibacterial agent reduced the fever immediately, and the inflammatory response was normalized. Three-dimensional CT scans of the chest revealed an afferent vessel from the pulmonary artery in the sequestered lung segment and reflux of the pulmonary vein into the innominate vein (Figure 2). The diagnosis was pulmonary sequestration accompanied by partial anomalous pulmonary venous return. VATS for pulmonary sequestration was performed because of the risk of secondary infection. Ports were inserted via a 3-cm skin incision in the midaxillary line in the fourth intercostal space, 1-cm incisions in the anterior axillary line in the fifth intercostal space, and 1-cm incisions in the posterior axillary line in the seventh intercostal space. The entire operation was carried out with a monitor, without use of a rib retractor. The sequestered lung segment was covered with visceral pleura and was thus diagnosed as an extralobar sequestration. As seen in the preoperative 3-dimensional CT findings, an afferent vessel from the left main pulmonary artery and reflux of the pulmonary vein into the innominate vein were observed by ablation of the hilar region. The afferent vessel of the pulmonary artery and the pulmonary vein were severed with an endoscopic linear cutter. In addition, the sequestered lung segment was dissected. The operation lasted 3 hours 20 minutes, and 15 g of blood was lost. No complications were observed and the patient was discharged from the hospital 4 days after the operation.
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Extralobar sequestration is believed to mostly affect male subjects and occurs between the diaphragm and lower lobe of the lung. The aberrant arteries commonly originate in the descending aorta or abdominal aorta. Previous studies have demonstrated that afferent vessels from the pulmonary artery are observed in 5% of all cases of extralobar sequestration. Most vein refluxes from extralobar sequestrations involve the hemiazygos vein, of which 80% flow into systemic veins, including the azygos vein and the inferior vena cava.1,2
The present patient had a sequestered lung segment in the left upper lobe without aberrant arteries, which is a rare clinical condition. The patient also had the complication of a pulmonary cyst. Therefore, we suspected that the patient had congenital cystic adenomatoid malformation (CCAM). CCAM is often observed in patients with extralobar sequestration. Previous studies have demonstrated that malignant diseases often develop in patients with CCAM, and thus early excision is recommended.3
As in the current case, it is difficult to firmly define the clinical manifestations of pulmonary sequestration. Some cases have not conformed to the original definition of pulmonary sequestration as proposed by Pryce.4 Sade,2 Clements,5 and their associates have suggested that there might be varying combinations of malformation, which they term the sequestration spectrum or pulmonary malinosculation spectrum. As in the current case, pulmonary sequestration is generally characterized by the diversity of afferent and reflux vessels and the existence of varying combinations of associated malformations.
Recently developed thoracoscopic surgery techniques allowed us to safely perform a lobectomy. Pulmonary sequestration is considered to be a benign disease, and VATS is indicated for its treatment.3,6 However, attention needs to be paid to abnormal structures such as aberrant arteries and associated malformations. Detailed preoperative identification of aberrant arteries is essential for safe thoracoscopic surgery. Three-dimensional CT is extremely useful for the identification of aberrant arteries, afferent vessels, and reflux vessels, and invasive angiography is not required given current 3-dimensional CT technology. In the present case, 3-dimensional CT without angiography was implemented to precisely characterize the afferent and reflux vessels and to perform VATS safely. VATS is indicated for pulmonary sequestration, and carrying out this operation requires the skills necessary for VATS lobectomy, such as ablation or incision of the pulmonary vessels.
References
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