Ann Thorac Surg 1997;63:1171-1173
© 1997 The Society of Thoracic Surgeons
Case Report
Simultaneous Discovery of Bilateral Intralobar and Extralobar Pulmonary Sequestrations
Thierry Jeanfaivre, MD,
Moncef Afi,
Philippe L'hoste, MD,
Edmond Tuchais, MD
Services de Pneumologie et Radiologie, Centre Hospitalier et Universitaire d'Angers, Angers Cedex, France
Accepted for publication November 30, 1996.
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Abstract
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We describe the rare case of bilateral bronchopulmonary sequestrations, one intralobar and one extralobar, in a 22-year-old man. The coexistence of two forms of separated sequestrations with a common vascularization support the theory that intralobar as well as extralobar sequestrations are congenital malformations.
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Introduction
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Since the first description by Rektorzick in 1861, 7 cases of bilateral sequestrations have been reported, but only Rosado-de-Christenson and associates [1] mentioned an observation of bilateral intralobar and extralobar pulmonary sequestrations, unfortunately without detail. We describe a case that is unusual in its radiologic and anatomic presentation.
Fifteen days after tonsillitis treated with penicillin V, a 22-year-old man with no previous history of respiratory symptoms was hospitalized with dry cough, dorsal transverse chest pains, and fever (39°C). The standard chest roentgenograms (Figs 1, 2
) showed a rounded density on the left, located at the rear of the heart, which contained an air-fluid level accompanied by a pleural effusion. The right cardiophrenic angle presented an additional heterogeneous density. The presence of two important symmetric masses, located in the inferior and internal rib and vertebral grooves on either side of the spine, was confirmed by computed tomographic scan (Fig 3). These two densities had a diameter of 8.5 cm. They contained multiple small cysts filled with fluid. The left opacity contained fluid and air. A bronchoscopic examination showed that the airways were normal. However, the bronchographic examination revealed that the inferior left lobes were pushed back, but without anomaly in the distribution of the airways. Two abnormal arteries were detected by aortography. On the forward edge of the aorta, at the level of the twelfth thoracic vertebra, just above the celiac trunk, a voluminous abnormal artery extended toward the lower density located on the right, and branched out when it reached the inferior pole (Fig 4). It extended over the external superior edge of this opaque area by means of smaller branches, which merged with a pulmonary venous branch. Another thin abnormal artery was observed (see Fig 4) that originated from the former abnormal artery and extended toward the lower left density. The aortography also indicated an abnormal vascularization of these two structures by small branches originating from the inferior phrenic artery, itself originating from the initial portion of the celiac trunk (Fig 5). The venous return from the left thoracic structure occurred through a subphrenic vein toward the coronary vein, which joined the splenic vein and the portal vein.
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Fig 3. . Lung computed tomographic scan using a mediastinal window: arrows show pulmonary sequestrations.
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Fig 4. . Selective arteriogram of the additional artery located above the celiac trunk. (1 = main artery feeding the right sequestration; 2 = secondary artery of the left sequestration.)
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Fig 5. . Selective arteriogram of the celiac trunk. (1 = celiac trunk; 2 = secondary bifid artery feeding the right sequestration; 3 = main artery feeding the left sequestration.)
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The two malformations were removed by two successive operations. In the first, the left thoracic structure was removed through a posterolateral thoracotomy along the superior edge of the sixth rib on the left-hand side. The excised mass was covered by an inflammatory serous membrane. The lesion was completely encapsulated and easily separable from the lung. From its lower pole extended a transdiaphragmatic pedicle, which included vessels: two arteries and one vein without connection to the adjacent lung. The vessels were ligated before they were severed. Two weeks later, the second structure was excised through a posterolateral thoracotomy along the superior edge of the seventh rib on the right side. The malformation was partly embedded within the base of the inferior lobe. It could not be dissociated from it. Resection of some healthy parenchyma was necessary. The mass was linked to the costodiaphragmatic sinus and to the mediastinum by a large pedicle, which contained two feeding arteries. The arteries were sectioned off after ligation. The pedicle was connected to the esophagus by adhesions without communication. No postoperative complications occurred after the two operations.
Macroscopic examination of the two surgically removed masses showed that they were composed of multicystic cavities, confirmed by microscopic examination. The left malformation was an extralobar sequestration, and the right mass was an intralobar sequestration.
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Comment
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Our observation is remarkable in many ways. Although extralobar sequestrations may be asymptomatic in most cases, the extralobar sequestration observed in this patient presented an air-fluid level, which was indicative of suppuration. Furthermore, in spite of the fact that an intralobar sequestration is usually discovered because of infectious signs [2], the right intralobar sequestration was only responsible for pain in our patient. Reports of transverse chest pains are not common in the literature, and such pains in this patient may be due to the exceptional bilateral character of the malformation.
The most unusual finding is the discovery of a common vascularization for two well-separated sequestrations. Our report reopens the discussion on the etiopathogenesis of pulmonary sequestrations. More particularly, an extralobar sequestration is usually considered to be of congenital origin [3]. However, the origin of intralobar sequestrations remains controversial. According to some authors, the latter could be of congenital origin just as are the extralobar sequestrations [4]. Other authors consider intralobar lesions to be acquired subsequent to various processes, such as localized infection [5], bronchial obstruction, and occlusion of the pulmonary circulation [6]. Several others reported arguments confirming the hypothesis of an acquired origin from the inhalation of a foreign body [7] or from a mycetoma [8]. However, several authors reported combined sequestrations, both intralobar and extralobar, and suggested that they originated from a common initial anomaly [4]. In our patient, the coexistence of two forms of separated sequestrations with a common vascularization supports the hypothesis that these sequestrations have a common embryologic origin.
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Footnotes
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Address reprint requests to Dr Jeanfaivre, Service de Pneumologie, Centre Hospitalier et Universitaire d'Angers, 49033 Angers Cedex 01, France.
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References
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- Rosado-de-Christenson ML, Frazier AA, Stocker JT, Templeton PA. Extralobar sequestration:radiologic-pathologic correlation. Radiographics 1993;13:425–41.[Abstract]
- Caron J, Bennet J, Pinet F, Caron-Poitreau C. Les séquestrations pulmonaires intra-lobaires (20 observations). Poumon Coeur 1970;4:423–43.
- Boyden FA. Bronchogenic cysts and the theory of intralobar sequestration. New embryologic data. J Thorac Surg 1958;35:604–16.
- Blesovsky A. Pulmonary sequestration. A report of an unusual case and a review of the literature. Thorax 1967;22:351–7.[Abstract/Free Full Text]
- Gebauer PW, Mason CB. Intralobar pulmonary sequestration associated with anomalous pulmonary vessels. A nonentity. Dis Chest 1959;35:282–8.
- Stocker JT, Malezak HT. A study of pulmonary arteries. Relationship to intralobar pulmonary sequestration. Chest 1984;86:611–5.[Abstract/Free Full Text]
- Kushner C. Case 48-1983. N Engl J Med 1983;309:1374–81.[Medline]
- Uppal MS, Kohman LJ, Katzenstein AA. Mycetoma within an intralobar sequestration. Evidence supporting acquired origin for this pulmonary anomaly. Chest 1993;103:1627–8.[Abstract/Free Full Text]
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Abstract
Introduction
