Interact CardioVasc Thorac Surg 2008;7:527-528. doi:10.1510/icvts.2007.170225
© 2008 European Association of Cardio-Thoracic Surgery
Case report - Thoracic general |
Left upper lobe pulmonary sequestration
Pascal Berna,
João Carlos das Neves Pereira,
Jean-François Coté and
Marc Riquet*
Thoracic Surgery Department, Georges Pompidou European Hospital, 20-40 rue Leblanc, 75015 Paris, France
Received 15 October 2007;
received in revised form 18 December 2007;
accepted 1 February 2008
*Corresponding author. Tel.: +33-1-56093450; fax: +33-1-56093380.
E-mail address: marc.riquet{at}egp.aphp.fr (M. Riquet).
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Abstract
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A 64-year-old non-smoking woman with a left upper lobe mass underwent a wedge resection by video-thoracoscopic approach. Intraoperative frozen section examination was unable to rule out a neoplastic disease and a lobectomy was performed. Microscopic analysis showed a typical pulmonary sequestration pattern including arteries with elastic layers in a systemic fashion. Pulmonary sequestration is a development lung disease: upper lobe location is considered uncommon and much controversy exists concerning this form which may be underrated.
Key Words: Pulmonary sequestration
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1. Introduction
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Pulmonary sequestration (PS) is a development lung disease defined as non-functioning pulmonary tissue lacking normal communication with the bronchial tree and supplied by a systemic artery. Two forms of sequestration are recognized [1]: extralobar, which is separated from the lung tissue by a separate lining of pleura, and intralobar, which is embedded in the normal lung sharing with it a common pleural investment. Intralobar sequestration is virtually absent in infants, rarely associated with malformations (6–12%), and rarely discovered in perinatal autopsies [1–3]. Therefore, some workers consider that it is an acquired condition in which the ‘aberrant’ artery is a dilated systemic collateral to a focus of infection [4]. Intralobar sequestrations are usually located in the lower lobes. Savic and colleagues [2] mentioned eight upper lobe PS out of 400 published cases, and only two were on the left. Our purpose was to question again this entity about a particular left upper lobe lesion masquerading a lung tumor.
The patient, a 64-year-old non-smoking woman, without past history of hemoptysis nor infectious pulmonary disease, presented with a non-productive cough. Chest radiograph demonstrated a left upper lobe mass, confirmed by computed tomography imaging. The mass was not present on a chest radiograph performed three years previously. Bronchoscopy, biopsy, and blind parenchymal biopsies were normal. The patient underwent a wedge resection by video-thoracoscopic approach for a suspicious lesion. Macroscopic features showed a round reddish lesion confined to the upper lobe, without sharp limit between them. Visceral pleura was intact. A small adhesion to the mediastinum was released. Intraoperative frozen section examination showing glandular-like cavities was unable to rule out a neoplastic disease and a lobectomy with complete media-stinal lymphadenectomy was performed via a standard muscle sparing thoracotomy. Resection was easy, except for the necessity of ligating a large caliber bronchial artery supplying the lobe. Postoperative course was uneventful, and the patient was faring well two years later. Microscopic analysis showed a pulmonary parenchyma without malignancy, but typical PS pattern with no communication between the sequestered parenchyma and the normal bronchial tree; large arterial branches were present with elastic layers in a systemic fashion (Fig. 1). The first chest radiograph was reviewed and actually showed a slight ‘honeycombing’ pattern of the upper lobe parenchyma retrospectively suggesting a pre-existing lesion. Reviewing the computed tomography contrast enhanced study permitted to follow a large systemic bronchial artery supplying the lesion (Fig. 2), which had been overlooked.
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Fig. 1. (a) Section of well-defined intralobar sequestration showing an anomalous artery (arrows) in the middle of cystic lung parenchyma changes with marked acute and chronic inflammation (Hematoxylin Eosin and Safran, x100). (b) Detail of the systemic artery's wall type.
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Fig. 2. Computed tomography: the systemic arterial supply is a large systemic bronchial artery (arrow head) running along the left upper bronchus and entering the lesion (arrow).
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2. Comment
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We suggest that the lesion we report is one of the extremely rare left upper lobe PS (0.5% [2]) ever observed. Presenting as an acquired condition, although its congenital nature cannot be definitely excluded, it illustrates the imprecise nosological boundaries of this entity. Chronic pneumonia of the upper lobes, histologically similar to intralobar PS is not unusual [3]. Systemic hypervascularization originating from the chest wall and parietal pleura and supplying the disease via aquired pleural adhesions is not uncommon. A unique artery such as the one reported by Hattori and colleagues [5] originating from the left internal thoracic artery is an exceptional finding, however, not precluding an acquired disease. From a strictly anatomical and congenital point of view, systemic arteries other than the bronchial arteries available to parasitize a chronic upper lobe sequestration are lacking [3]. These arteries do not appear aberrant and may be overlooked. All that probably explains why upper lobe PS may remain unrecognized and accounts for the rare occurrence of the disease. However, the frequency may be underrated, most cases being probably unpublished due to difficulty demonstrating this entity according to academic discussion.
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References
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- Corrin B. Pathology of the lungs. London: Churchill Livingstone; 2005.
- Savic B, Birtel FJ, Tholen W, Funke HD, Knoche R. Lung sequestration: a report of seven cases and review of 540 published cases. Thorax 1979;34:96–101.[Medline]
- Stocker JT, Dehner LP. Acquired neonatal and pediatric disease. Intralobar sequestration. In: Dail DH, Hammar SP, Pulmonary Pathologie, 2nd ed. New York: Springer Verlag; 1993:223–227.
- Stocker JT, Malczak HT. A study of pulmonary ligament arteries. Relationship to intralobar sequestration. Chest 1984;86:611–615.[CrossRef][Medline]
- Hattori Y, Negi K, Takeda I, Iriyama T, Sugimura S, Watanabe K. Intrapulmonary sequestration with arterial supply from the left internal thoracic artery: a case report. Ann Thorac Cardiovasc Surg 2000;6:119–121.[Medline]
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Abstract
1. Introduction