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Congenital Intralobar Pulmonary Sequestration with Cystic Adenomatoid Malformation

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Congenital Intralobar Pulmonary Sequestration with Cystic Adenomatoid Malformation Journal of Perinatology (2009) 29, 175–177 r 2009 Nature Publishing Group All rights reserved. 0743-8346/09 $32 www.nature.com/jp IMAGING CASE REPORT Congenital intralobar pulmonary sequestration with cystic adenomatoid malformation TE Herman and MJ Siegel Mallinckrodt Institute of Radiology, Washington University School of Medicine, St Louis, MO, USA Journal of Perinatology (2009) 29, 175–177; doi:10.1038/jp.2008.90 cystic adenomatoid malformation with intermediate-type cysts between types 1 and 2. Intralobar sequestration (ILS) refers to nonfunctioning lung Case presentation tissue with anomalous arterial supply from the aorta, venous A 3000 g infant was born at 39 weeks gestation to a gravida 2, para 2 mother after a pregnancy complicated by an abnormal fetal sonogram that was reported to demonstrate a large cystic thoracic mass. The baby was delivered by elective repeat C-section and had Apgar scores of 8 at 1 min and 9 at 5 min. The infant was not in distress. An initial plain chest radiograph (Figure 1), abdominal– thoracic sonogram (Figure 2) and a multidetector chest computed tomography (CT) and CT angiogram (Figure 3) were then performed. Denouement and discussion The chest radiograph shows subtle lucency in the left lower lobe and hyperexpanded left lung. The sonogram showed an echogenic mass with feeding artery. CT confirmed the presence of an intrapulmonary, hypervascular cystic and solid mass in the medial posterior left lower lobe. The lesion receives arterial blood supply from an anomalous vessel directly arising from the thoracic aorta Figure 1 Frontal supine chest radiograph with subtle lucency at the left base in a slightly hyperexpanded left lung. and has drainage through an anomalous vein into the azygos vein. The air-filled cystic component of the lesion is peripherally located and at the interface with the more normal lung. The remainder of the lesion is not aerated, but is surrounded by normally aerated left lower lobe, medially, laterally and superiorly. Because of the risk of infection and malignant degeneration, which is associated with the presumed cystic adenomatoid elements in this lesion, the lesion was resected. Surgical resection demonstrated a cystic and solid mass in the left lower lobe with blood supply from a 1-cm diameter artery arising directly from the thoracic aorta with venous drainage into the azygos vein. This complex mass was within the left lower lobe and within the visceral pleural. Pathological examination of the specimen confirmed a sequestration and associated congenital Correspondence: Dr TE Herman, Mallinckrodt Institute of Radiology, St Louis Children’s Hospital, Washington University School of Medicine, 510 South Kingshighway Blvd, St Louis, MO 63110, USA. Figure 2 Axial chest sonogram of the lower left paraspinal region. An E-mail: [email protected] anomalous artery (arrowhead) from the thoracic aorta (A) is seen extending to a Received 30 April 2008; accepted 13 May 2008 left paraspinal echogenic mass (arrow). Congenital intralobar pulmonary sequestration TE Herman and MJ Siegel 176 Figure 3 (a) Axial CT angiogram demonstrates an anomalous vein (curved arrow) draining to a left paraspinal hypervascular mass (straight arrow). The vein is directly anterior to the descending thoracic aorta (A). (b) Axial image at a slightly more caudal level showing the anomalous vein (curved arrow) draining toward the right side. A large artery arises from the aorta (straight arrow) to supply the enhancing sequestration (S) in the left paraspinal region. (c and d) Axial CT images at lung windows showing cystic and solid components in the sequestration in the medial posterior left lower lobe.(e) Three-dimensional reconstruction of angiogram showing extensive vascularity in the left basilar sequestration with a large feeding artery (arrow) arising perpendicularly from the thoracic aorta (A) and an anomalous vein (curved arrow) draining into the azygos vein (Z). CT, computed tomography. Journal of Perinatology Congenital intralobar pulmonary sequestration TE Herman and MJ Siegel 177 drainage usually to pulmonary veins but occasionally to systemic malformation and were intermediate between type 1, in which the veins, the same pleural investment as normal lung and no or only cysts are greater than 3 cm, and type 2 in which the cysts vary a miniscule patent bronchial connection.1,2 This lesion is between 0.5 and 3.0 cm.8 The presence of cystic adenomatoid differentiated from extralobar sequestration in which the portion of malformation within ILS is also usually type 2.9 lung tissue with abnormal arteriovenous connections is completely The initial detection of congenital pulmonary sequestrations or outside the visceral pleural and may be outside the thorax.2 other bronchopulmonary foregut malformations is currently often ILSs are very rare in newborns. In a series of 42 000 autopsies of by intrauterine sonography. Postnatal evaluation is best performed children who had died younger than 2 months of age, 150 cases of by multi-detector CT evaluation with intravenous contrast for CT extralobar sequestration were found, but not a single case of ILS.2 angiogram. This study provides the best evaluation of the vascular This led to speculation that ILSs were acquired and not congenital anatomy of the lesion and the appearance of the lesion itself, and lesions. However, ILS has been found in neonates, and ILS is a of the diaphragm and remainder of the lung.10 Although magnetic congenital malformation. resonance imaging can be performed without radiation and does ILSs, although rare in the neonate, are more common overall provide excellent vascular visualization, it does not provide the than extralobar sequestrations, comprising 75% of all report cases same high-quality evaluation of the pulmonary parenchymal part of pulmonary sequestrations.3 Pulmonary sequestrations are of the lesion.11 complex malformations, many of which do not easily fit into In addition to the concern for recurrent infections in ILS, there current nomenclature easily.4 There are six features that must is also a currently recognized risk in cystic adenomatoid be evaluated to characterize a particular lesion. These include malformation or congenital lung cysts for development of (a) bronchial anatomy, (b) arterial supply, (c) venous drainage, pleuropulmonary blastoma.8 Because of these risks, surgical (d) upper gastrointestinal tract, (e) diaphragm and (f) other resection or very close follow-up of all congenital lung cysts is pulmonary anomalies. recommended. An ILS has bronchial atresia or miniscule patency of the bronchus, accommodating only a tiny probe, to that segment. However, aeration can be seen in sequestration and is usually References attributed to a tiny patent bronchus or to collateral air drift.1 The arterial supply is from systemic arteries, 74% directly from the 1 Takahashi M, Ohno M, Mihara K, Matsuura K, Sumiyoshi A. Intralobar pulmonary thoracic aorta, 12% from the abdominal aorta and the remainder sequestration with special emphasis on bronchial communication. Radiology 1975; 5 114: 543–549. from other arteries such as the subclavian artery. The aortic vessel 2 Stocker JT. Sequestrations of the lung. Semin Diagn Pathol 1986; 3: 106–121. is usually large, varying in size from 1 mm to 1.5 cm in diameter. 3 Thilenius OG, Ruschhaupt DG, Reploge RL, Bharati S, Herman T, Arcilla RA. Spectrum Atherosclerotic changes are common in the anomalous artery in of pulmonary sequestration: association with anomalous pulmonary venous drainage ILS, even in young patients.6 The pulmonary venous drainage is in infants. Pediatr Cardiol 1983; 4: 97–103. predominantly to the left atrium in 95% of cases.5 If the drainage is 4 Langston C. Intralobar sequestration, revisited. Pediatr Dev Pathol 2003; 6: 23. 5 Savic B, Birtel FJ, Tholen W, Funke HD, Knoche R. Lung sequestration: report of seven to the system veins (4.25% of cases), it occurs primarily to the cases and review of 540 published cases. Thorax 1979; 34: 96–101. inferior vena cava and to the azygos vein, as in this patient. 6 Frazier AA, Rosado de Christenson ML, Stocker TJ, Templeton PA. Intralobar Communication with the esophagus occurs in some sequestrations sequestration: radiologic pathologic correlation. Radiographics 1997; 17: 725–745. and creates a distinctive and rare lesion and a very complex type of 7 Chadha R, Sing D, Kathuria P, Sharma S, Choudhur SR, Jain M. Congenital cystic bronchopulmonary foregut malformation. Diaphragmatic hernias, adenomatoid malformation associated with ipsilateral eventration of the diaphragm. defects and eventration probably occur more frequently with Indian J Pediatr 2006; 73: 832–834. 7 8 Zangwill BC, Stocker JT. Congenital cystic adenomatoid malformation within an extralobar sequestrations than intralobar. extralobar pulmonary sequestration. Pediatr Pathol 1993; 13: 309–315. Cystic changes are common in ILS. In older patients, these are 9 Samuel M, Burge DM. Management of antenatally diagnosed pulmonary sequestration often sequelae of infection. Cystic changes in extralobar associated with congenital cystic adenomatoid malformation. Thorax 1999; 54: sequestrations are present in 25% of cases and are almost always 701–706. type 2 congenital cystic adenomatoid malformations. In ILS, causes 10 Lee EY, Siegel MJ, Sierra LM, Foglia RP. Evaluation of angioarchitecture of pulmonary sequestration in pediatric patients using 3D MDCT angiography. AJR 2004; 183: of cystic lesions include bronchiectasis and cystic adenomatoid 183–188. 5 malformation, which can be unicystic or polycystic. In this 11 Choudry R, Salvatore M, Hurrie J, Grewal H. Preoperative magnetic resonance imaging patient, the cystic lesions were related to a cystic adenomatoid for pulmonary sequestration. J Pediatr Surg 2006; 41: 872–874. Journal of Perinatology.
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