Ann Thorac Surg 2009;87:1939-1941. doi:10.1016/j.athoracsur.2008.11.008
© 2009 The Society of Thoracic Surgeons
Case Reports
Simultaneous Resection of Bilateral Intralobar and Extralobar Pulmonary Sequestrations With Video-Assisted Thoracoscopic Surgery
Yoshiyuki Yamamura, MD,
Yasuhiro Hida, MD, PhD*,
Kichizo Kaga, MD, PhD,
Masaya Kawada, MD, PhD,
Hiroto Niizeki, MD, PhD,
Masaomi Ichinokawa, MD,
Satoshi Kondo, MD, PhD
Department of Surgical Oncology, Hokkaido University Graduate School of Medicine, Hokkaido, Japan
Accepted for publication November 3, 2008.
* Address correspondence to Dr Hida, Department of Surgical Oncology, Hokkaido University Graduate School of Medicine, North 15, West 7, Kita-ku, Sapporo, Hokkaido, 060-8648, Japan (Email: yhida{at}med.hokudai.ac.jp).
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Abstract
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The term pulmonary sequestration is applied to a pulmonary lobe or portion of a lobe that is supplied by an anomalous systemic artery and drain either into the systemic or pulmonary veins. The conditions are divided into intralobar pulmonary sequestration, in which the sequestration is situated inside the visceral pleura of a normal lobe, and extralobar sequestration, in which the sequestration is surrounded by its own pleura. Most sequestrations are unilateral; bilateral sequestrations are rare. We report the case of a synchronous bilateral intralobar and extralobar pulmonary sequestrations resected simultaneously with video-assisted thoracoscopic surgery.
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Introduction
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Bilateral pulmonary sequestration is extremely rare, and the traditional surgical approach has been sequential resections with staged thoracotomies. We report the case of a man who underwent synchronous resection with video-assisted thoracoscopic surgery (VATS).
A 41-year-old man visited a hospital with a low-grade fever and cough. A chest roentgenogram showed a round density of the right lower lung field and a similar one of the left lower lung field (Fig 1). A computed tomographic (CT) scan of the chest showed bilateral, oval, soft-tissue mass on the left and right lower lobes, with a possible bridging tunnel and an aberrant feeding vessel from the thoracic descending aorta (Fig 2). Computed tomographic scan with three-dimensional reconstructions also showed two aberrant arteries arising from the thoracic aorta. The upper artery (measuring 5 mm in diameter) supplied the right pulmonary sequestration, and the lower artery (measuring 1 mm in diameter) supplied the left pulmonary sequestration (Fig 3). The plan was to resect both sequestrations simultaneously with synchronous VATS. If the procedure were converted to open thoracotomy during VATS for the right lung, we would have performed staged thoracotomies for the left. The patient was initially placed in the right lateral decubitus position. The left pulmonary sequestration had adhered to the lower lobe and was sharply dissected from the lower lobe. We considered the left pulmonary sequestration to be an extralobar pulmonary sequestration. A small artery, coming from the thoracic aorta, was found and divided. The left pulmonary sequestration was connected to the right pulmonary sequestration. Resection of the left sequestration was performed. There was a bronchus between the left and the right sequestration. It was divided with a linear stapler in the mediastinum. No drainage vein was identified on the left side. A single 28-French chest tube was placed, and the incisions were closed in layers. The patient was then turned to the left lateral decubitus position. Because of massive adhesion, the right sequestrum could not be clearly differentiated from the right lower lobe. The right lower lobe was partially resected with the right sequestration. Another single 28-French chest tube was placed, and the incisions were closed in layers. Both chest tubes were removed 2 days later and the patient was discharged home on postoperative day 10. Final pathology revealed that the left malformation was an extralobar sequestration and the right mass was an intralobar sequestration, and there was a bronchial structure connecting the sequestrations (Fig 4).
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Fig 1. A chest roentgenogram showed a rounded density of the right lower lung field and similar one of the left lower field.
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Fig 2. Computed tomographic scan of the chest showing bilateral, oval, soft-tissue masses on left and right lower lobes, with a possible bridging tunnel and an aberrant feeding vessel from the thoracic descending aorta.
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Fig 3. Computed tomographic scan with three-dimensional reconstruction showed two aberrant arteries arising from the thoracic aorta. The upper artery (white arrow), measuring 5 mm in diameter, supplied the right pulmonary sequestration and the lower artery (black arrow), measuring 1 mm in diameter, supplied the left pulmonary sequestration.
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Fig 4. Pathology revealed that the left malformation was an extralobar sequestration and the right mass was an intralobar sequestration, and that there was a bronchial structure connecting the sequestrations (arrows).
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Comment
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The clinical feature, evaluation, and management of pulmonary sequestration have been well described in the literature. We believe that only 20 bilateral cases have been reported to date. There have been four reported cases of mixed bilateral pulmonary sequestration (one intralobar and one extralobar); all cases had a right intralobar and a left extralobar pulmonary sequestration, as in our case [1–4].
In the case of an intralobar pulmonary sequestration, venous drainage is made through the pulmonary veins; with an extralobar pulmonary sequestration, venous drainage is made through the systemic veins. Nicolette and associates [5] reported that in many cases of extralobar pulmonary sequestration, venous drainage is through the azygos vein. In our case, we were unable to identify a source of venous drainage in the left-side sequestration. From this, we considered that the right intralobar pulmonary sequestration initially developed, then part of this sequestration migrated into the left pleural cavity through the mediastinal space.
The most common cause of morbidity in pulmonary sequestration is infection, resulting from the air passage that may develop between the normal lung parenchyma and the sequestered area. If both sides are infected and are causing symptoms, it is clear that both sides should be resected; however, if only one side is symptomatic, a decision must be made regarding bilateral resection.
To date, there have been five reported cases of a synchronous bilateral resection. Morse and associates [6] reported a single case of a synchronous video-assisted resection of bilateral intralobar pulmonary sequestration. In our case, we performed a synchronous VATS approach to bilateral intralobar and extralobar pulmonary sequestrations. More recently, surgical resection of pulmonary sequestration has been accomplished with the VATS approach [7, 8]. Lobectomy, sequestrectomy, and wedge resections have all been described with an excellent result and a small number of conversions to a thoracotomy.
This unique case of bilateral pulmonary sequestrations demonstrated the feasibility of a single-setting, sequential VATS approach to minimize postoperative pain and limit multiple or prolonged hospitalizations.
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References
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- Spinella PC, Strieper MJ, Callahan CW. Congestive heart failure in a neonate secondary to bilateral intralobar and extralobar pulmonary sequestrations Pediatrics 1998;101:120-124.[Free Full Text]
- Pendse P, Alexander J, Khademi M, Groff DB. Pulmonary sequestration, coexisting classic intralobar and extralobar types in a child J Thorac Cardiovasc Surg 1972;64:127-131.[Medline]
- Jeanfaivre T, Afi M, L'hoste P, Tuchais E. Simultaneous discovery of bilateral intralobar and extralobar pulmonary sequestrations Ann Thorac Surg 1997;63:1171-1173.[Abstract/Free Full Text]
- Trudel JA, Lemire G, Rabbat AG. Bilateral sequestrations of different type with mirror-image vascularization Chest 1977;72:256-257.[Abstract/Free Full Text]
- Nicolette LA, Kosloske AM, Bartow SA, Murphy S. Intralobar pulmonary sequestration: a crinical and pathological spectrum J Pedia Surg 1993;28:802-805.
- Morse CR, Ishitani MB, Cassivi SD. Video-assisted resection of bilateral intralobar pulmonary sequestrations J Thorac Cardiovasc Surg 2006;131:917-918.[Free Full Text]
- Wan IY, Lee TW, Sihoe AD, Ng CS, Yim AP. Video-assisted thoracic surgery lobectomy for pulmonary sequestration Ann Thorac Surg 2002;73:639-640.[Abstract/Free Full Text]
- Sakuma T, Sugita M, Sagawa M, Ishigaki M, Togo H. Video-assisted thoracosopic wedge resection for pulmonary sequestration Ann Thorac Surg 2004;78:1844-1845.[Abstract/Free Full Text]
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Abstract
Introduction
