Pédiatrie and Developmental Pathology 17, 55-58, 2014 DOI: 10.2350/13-08-1371-CR.1 © 2014 Society for Pédiatrie Pathology
Bilateral Intralobar Sequestration of the Lung with a Bridging Isthmus: Pathologic and Radiologie Findings RYUJI FUKUZAWA/* KOJI KOMORI,^ TATSUO KOHNO,^ SEIICHI HIROBE,^ AND JUN MIYAUCHI^ ^Department of Pathology, Tokyo Metropolitan Children's Medical Center, 2-8-29 Musashidai, Fuchu, Tokyo 183-8561, Japan ^Department of Surgery, Tokyo Metropolitan Children's Medical Center, 2-8-29 Musashidai, Fuchu, Tokyo 183-8561, Japan ^Department of Radiology, Tokyo Metropolitan Children's Medical Center, 2-8-29 Musashidai, Fuchu, Tokyo 183-8561, Japan ''Department of Pathology and Laboratory Medicine, Tokyo Dental College Ichikawa General Hospital, 5-11-31 Sugano, Ichikawa 272-8513, Chiba, Japan Received August 28, 2013: accepted October 20, 2013; published online October 21, 2013.
ABSTRACT A 4-year-old boy presented with pneumonia. Computed tomography demonstrated a multicystic mass at the posteromedial bilateral lower lobe segments, which were connected by a bronchus behind the heart. Enhanced computed tomography revealed that an anomalous artery arose from the left gastric artei^ and inserted into the left sequestrated lung and branched to the right one. The diagnosis of bilateral intralobar sequestration (ILS) with a bridging isthmus was made. After removal of the bilateral ILS, radiologie and pathologic approaches were undertaken to reconstruct the vascular and bronchial architectures. The following observations were made: (1) histologically, the region near the anomalous artery insertion site contained bronchial structures, which looked like an ectopic pulmonary hilus. This bronchial structure was continuously observed in the isthmus and its opening of the right sequestrated lung; (2) radiologically, the shape and course of the bronchi within the ILS indicated a distinct bronchial origin that arose from the pulmonary hilus-like structure, and the anomalous artery that ran along with those bronchi, resembled a pulmonary artery. These features suggested that this bilateral ILS might have originated from an accessory lung tissue. Key words: congenital cystic adenomatoid malformation, congenital pulmonary airway malformation, lung bud, pulmonary development
INTRODUCTION Pulmonary sequestration is a nonflinctioning lung tissue that does not communicate with the tracheobronchial tree irrespective of the presence or absence of apparent bronchial atresia within the normal pleural investment * Corresponding author, e-mail: [email protected]
and that possesses an anomalous artery supplied from the systemic artery [1]. Pulmonary sequestration is classified into 2 types, depending on its extrapulmonary or intrapulmonary location. Extralobar sequestration of the lung (ELS) is an isolated mass of pulmonary tissue with its own pleural investment separated from the normal lung. It is thought to arise from an accessory lung bud as an outpouching of the foregut. separated from the normally developing lung [1]. On the other hand, intralobar sequestration of the lung (ILS) is a portion of the lung tissue within the normal pleural investment [1] found in the posterior, medial, and basal segments of the lower lobes. It was previously postulated to be an acquired anomaly resulting from the obliteration of bronchial connection from the main bronchi by repeated or chronic infection ([1,2]. However, recent studies have inferred a congenital origin for the condition [3,4]. Bilateral ILS with a bridging isthmus represents a variant of ILS whose left and right sequestrated lungs are connected by a communicating tunnel covered by visceral pleura [5]. Horseshoe lung is a similar condition in which both lungs are fused at the base by an isthinus of lung parenchyme behind the heart. However, horseshoe lung is distinct from bilateral ILS with a bridging isthmus in that the arterial supply to the isthmic part branches from the pulmonary artery and otherwise lacks aberrant arteries. To our knowledge, only 2 cases have been reported [5,6]. We report the vascular and bronchial architectures in a case of bilateral ILS with a bridging isthmus and discuss the pathogenesis. CLINICAL HISTORY A 4-year-old boy presented with cough and fever for 5 days and was admitted to a local hospital with the diagnosis of pneumonia. He was referred to our hospital because of abnormal chest shadows on imaging studies.
trunk to the lesion in the left lower lobe (Fig. IB). The aberrant artery was bifurcated immediately after insertion into the left lesion and then crossed to the right lesion via the connecting tunnel (Fig. IB). Venous return was documented through the right and left pulmonary veins. A gastrointestinal series gave no evidence of tracheoesophageal communication. The diagnosis of bilateral ILS was made, and bilateral resection (left lower lobectomy and right segmentectomy) was performed.
B Figure 1. Radiologie features of the bilateral intralobar sequestration (ILS) with a bridging isthmus. A. Chest computed tomography transverse section demonstrates multicystic lesions in the posterior, medial, and basal left and right lungs, which are connected by an air-filled bronchus (arrows). B. Three-dimensional reconstruction of the computed tomographic scan shows an aberrant artery (aal) branched from the left gastric artery via the celiac trunk and supplying the sequestration on the left. The aberrant artery is bifurcated immediately after insertion into the left ILS (aa2) and then crossed to the right ILS (aa3). Both lungs (right purple, left iight blue) are connected by a bronchus (light blue, arrow heads) derived from the left ILS.
He had no history of respiratory disease until this episode. Chest radiograph showed bilateral opacities occupying the left and right lower lobes. Computed tomography demonstrated a well-demarcated multicystic mass at the posteromedial bilateral lower lobe segments, which were connected by a bronchus behind the heart (Fig. lA). Contrast-enhanced computed tomography revealed an arterial supply from the left gastric artery via the celiac
56
R . FUKUZAWA ET AL.
METHODS AND PATHOLOGIC FINDINGS In order to examine the bronchial shape and course within the IES, we injected a radiopaque contrast medium into a bronchus in the isthmus before formalin fixafion. Both lungs contained sequestrated segments that were isolated from the normal bronchial tree but that could communicate by the connecting tunnel (Fig. 2B,C), confirming the diagnosis. The bronchi in the left sequestrated lung coursed laterally and upward from near the aberrant artery insertion site, and those in the right one ran from the bridging isthmus in a lateral direction (Fig. 2B,C). From the anomalous artery insertion site toward the hilus of the nonnal lung, the bronchi generally became smaller in diameter with increased branching in number and then formed acini (Fig. 2B,C). These features suggested that another bronchial system arose from near the anomalous artery insertion at the periphery of the lower lobe. Grossly, both lungs were connected by an isthmus covered by visceral pleura (Fig. 2A). Cut sections of both lungs showed a typical IES appearance: lesions consisting of lipid pneumonia with variably sized cystic changes were well-demarcated from the nonnal lung tissue. Histologically, the pulmonary parenchyma was distorted by chronic inflammation with lymphoid follicles. Dilated alveoli and bronchioles were filled with foamy macrophages. Both lesions contained congenital pulmonary airway malformation (CPAM) type II (Fig. 3A), but obstruction of a branch of the bronchus within the pulmonary parenchyma was identified only in the right sequestrated lung. The aberrant artery insertion site consisted of an elastic-type artery and bronchial tissue accompanied by cartilage, mucous glands, and smooth muscle, which looked like a pulmonary hilus (Fig. 3B). Stepwise serial sections taken from the communicating tunnel extending into the left sequestrated lung were all composed of similar bronchial tissue (Fig. 3C). These features were restricted to the region around the opening of the isthmus, and the normal segments of the left lower lobe demonstrated no significant pathologic change. DISCUSSION Since Pryce, who coined the term "sequestration," 1st hypothesized that traction by an anomalous branch of the aorta during segmentation of the developing lung resulted in separation from normal lung [7], a number of efiologic hypotheses have been proposed [2]. The hypothesis Gerle and colleagues [8] would seem to fit the findings in the
Figure 2. Pathologic features of tbe bilateral intralobar sequestration (ILS) witb a bridging isthmus. Gross feature of tbe bilateral ILS witb a bridging isthmus (arrow) (A). Radiographs showing tbe bronchial tree in botb tbe right (B) and left (C) sequestrated lungs. Note a catheter inserted into the aberrant artery near tbe stem bronchus-like structure. Both lungs contain tbe lesions isolated from tbe normal bronchial tree, confirming tbe diagnosis of ILS. Tbe shape and course of the bronchi demonstrate tbat tbe bronchial trees are not associated with the normal ones. Figure 3. Histologie features of the bilateral intralobar sequestration with a bridging isthmus. Congenital pulmonary airway malformation type II (A). Sections from near the aberrant artery insertion portion (B) and the bridging isthmus (C). The histologie features consist of bronchial tissue (br), cartilage (ca), gland (gl), and aberrant artery (aa), which resembles the pulmonary hilus. Hematoxylin and eosin, (A); elastic Van Gieson stain (B,C). Original magnification, x i o (A); x1.48 (B); X3.08 (C).
current case. They reported 3 cases of IES communicating with the gastrointestinal tract. They considered that ELS, IES, and sequestration that feature communication with the gastrointestinal tract share a common embryologie origin and suggested the term "congenital bronchopulmonary-foregut malfomiation" [8]. On histologie examination, our case showed that the region near the anomalous artery insertion site contained bronehial epithelial tissue with the presence of cartilage, smooth muscle, and bronchial glands, as well as elastic arteries branching from the anomalous artery (Fig. 3B,C). These structures resembled those making up the pulmonary hilus, and we therefore called them "pulmonary hilus-like structures." The following observations support this designation; (1) a bronchogram within the IES confirmed that the bronchi in both IESs were not derived from the normal bronchial tree, because the orientation of the bronchi was different from that of normal bronchi in the adjacent nornial lung (Fig. 2 B,C); (2) the aberrant elastic-type artery ran along the bronchi (Fig. 3B,C), which is characteristic of a pulmonary artery rather than a systemic artery. The question arises as to how such a hilus-like structure is formed. The anomalous artery has been
considered to be a remnant of a primitive aortic branch that originally had supplied the developing lung bud [9]. We speculate that persistence of the primitive aortic branch prevents regression of a pluripotent tissue, leading to development of an additional lung tissue that might eventually incorporate into the normal lung and form an ILS. In our case, we suggest that the lefr ILS initially developed from near the anomalous artery insertion portion where the hilus-like structure was histologically noted and that part of the lefr developing malformed lung tissue subsequently extended to the right side and gave rise to the bridging isthmus and the right IES. This pathogenetic assumption might be explained by the hypothesis that ILS originates from an aecessoty lung bud [8]. Recently, it has been thought that airway obstruction at bronchiolar formation levels during lung development (the psedoglandular phase: 7-17 weeks of gestation) consequently causes a parenchymal hamartomaous lesion referred to as CPAM [10]. In fact, Riedlinger and eollagues reported that bronchial atresia and CPAM were present in 82% and 91% of ILS, respectively [4]. Thus, the coexistence of bronchial atresia and CPAM in our BILATERAL ILS WITH BRIDGING ISTHMUS
57
case indicates that our bilateral ILS shares a common pathogenetic mechanism with conventional unilateral cases, supporting that ILS is a congenital anomaly. ACKNOWLEDGMENT This work was supported by Clinical Research Grants from the Tokyo Metropolitan Government and Grant-inAid for Scientific Research from the Ministry of Education, Culture, Sports, Science and Technology of Japan (25460427).
4.
5.
6.
7.
REFERENCES 1. Stocker JT, Maní H, Husain AN. The respiratory tract. In: Stocker JT, Dehner MD, Husain AN, eds. Stocker and Dehner's Pédiatrie Pathology. 3rd ed. Riverwoods, IL: Lippincott Williams & Wilkins, 2010;441-515. 2. Stocker JT, Malczak HT. A study of pulmonary ligament arteries: relationship to intralobar pulmonary sequestration. Chest 1984;86: 611-615. 3. Walford N, Htun K, Chen J, Liu YY, Teo H, Yeo GS. Intralobar sequestration of the lung is a congenital anomaly: anatomopatho-
58
R . FUKUZAWA ET AL.
8.
9.
10.
logical analysis of four cases diagnosed in fetal life. Pediatr Dev Pathol 2003;6:314-32I. Riedlinger WF, Vargas SO, Jennings RW, et al. Bronchial atresia is common to extralobar sequestration, intralobar sequestration, congenital cystic adenomatoid malfon^nation, and lobar emphysema. Pediatr Dev Pathol 2006;9:361-373. Kuo R, Shih SL, Huang JK. On the AJR digital viewbox: bilateral pulmonary sequestration with bridging isthmus in a boy with Williams syndrome. AJR Am J Roentgenol 2006;187:W133-W134. Cerruti MM, Marmolejos F, Caeciarelli T. Bilateral intralobar pulmonary sequestration with horseshoe lung. Ann Thorac Surg 1993;55:509-510. Pryce DM. Lower accessory pulmonary artery with intralobar sequestration of lung: a report of seven cases. J Pathol Bacteriol 1946;58:457^67. Gerle RD, Jaretzki A 3rd, Ashley CA, Berne AS. Congenital bronchopulmonary-foregut malformation: pulmonary sequestration communicating with the gastrointestinal tract. N Engl J Med 1968; 278:1413-1419. Ellis K. Fleischner lecture: developmental abnormalities in the systemic blood supply to the lungs. AJR Am J Roentgenol 1991; 156:669-679. Langston C. New concepts in the pathology of congenital lung malformations. Semin Pediatr Surg 2003;12:17-37.
Copyright of Pediatric & Developmental Pathology is the property of Allen Press Publishing Services Inc. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
Bilateral Intralobar Sequestration of the Lung with a Bridging Isthmus: Pathologic and Radiologie Findings RYUJI FUKUZAWA/* KOJI KOMORI,^ TATSUO KOHNO,^ SEIICHI HIROBE,^ AND JUN MIYAUCHI^ ^Department of Pathology, Tokyo Metropolitan Children's Medical Center, 2-8-29 Musashidai, Fuchu, Tokyo 183-8561, Japan ^Department of Surgery, Tokyo Metropolitan Children's Medical Center, 2-8-29 Musashidai, Fuchu, Tokyo 183-8561, Japan ^Department of Radiology, Tokyo Metropolitan Children's Medical Center, 2-8-29 Musashidai, Fuchu, Tokyo 183-8561, Japan ''Department of Pathology and Laboratory Medicine, Tokyo Dental College Ichikawa General Hospital, 5-11-31 Sugano, Ichikawa 272-8513, Chiba, Japan Received August 28, 2013: accepted October 20, 2013; published online October 21, 2013.
ABSTRACT A 4-year-old boy presented with pneumonia. Computed tomography demonstrated a multicystic mass at the posteromedial bilateral lower lobe segments, which were connected by a bronchus behind the heart. Enhanced computed tomography revealed that an anomalous artery arose from the left gastric artei^ and inserted into the left sequestrated lung and branched to the right one. The diagnosis of bilateral intralobar sequestration (ILS) with a bridging isthmus was made. After removal of the bilateral ILS, radiologie and pathologic approaches were undertaken to reconstruct the vascular and bronchial architectures. The following observations were made: (1) histologically, the region near the anomalous artery insertion site contained bronchial structures, which looked like an ectopic pulmonary hilus. This bronchial structure was continuously observed in the isthmus and its opening of the right sequestrated lung; (2) radiologically, the shape and course of the bronchi within the ILS indicated a distinct bronchial origin that arose from the pulmonary hilus-like structure, and the anomalous artery that ran along with those bronchi, resembled a pulmonary artery. These features suggested that this bilateral ILS might have originated from an accessory lung tissue. Key words: congenital cystic adenomatoid malformation, congenital pulmonary airway malformation, lung bud, pulmonary development
INTRODUCTION Pulmonary sequestration is a nonflinctioning lung tissue that does not communicate with the tracheobronchial tree irrespective of the presence or absence of apparent bronchial atresia within the normal pleural investment * Corresponding author, e-mail: [email protected]
and that possesses an anomalous artery supplied from the systemic artery [1]. Pulmonary sequestration is classified into 2 types, depending on its extrapulmonary or intrapulmonary location. Extralobar sequestration of the lung (ELS) is an isolated mass of pulmonary tissue with its own pleural investment separated from the normal lung. It is thought to arise from an accessory lung bud as an outpouching of the foregut. separated from the normally developing lung [1]. On the other hand, intralobar sequestration of the lung (ILS) is a portion of the lung tissue within the normal pleural investment [1] found in the posterior, medial, and basal segments of the lower lobes. It was previously postulated to be an acquired anomaly resulting from the obliteration of bronchial connection from the main bronchi by repeated or chronic infection ([1,2]. However, recent studies have inferred a congenital origin for the condition [3,4]. Bilateral ILS with a bridging isthmus represents a variant of ILS whose left and right sequestrated lungs are connected by a communicating tunnel covered by visceral pleura [5]. Horseshoe lung is a similar condition in which both lungs are fused at the base by an isthinus of lung parenchyme behind the heart. However, horseshoe lung is distinct from bilateral ILS with a bridging isthmus in that the arterial supply to the isthmic part branches from the pulmonary artery and otherwise lacks aberrant arteries. To our knowledge, only 2 cases have been reported [5,6]. We report the vascular and bronchial architectures in a case of bilateral ILS with a bridging isthmus and discuss the pathogenesis. CLINICAL HISTORY A 4-year-old boy presented with cough and fever for 5 days and was admitted to a local hospital with the diagnosis of pneumonia. He was referred to our hospital because of abnormal chest shadows on imaging studies.
trunk to the lesion in the left lower lobe (Fig. IB). The aberrant artery was bifurcated immediately after insertion into the left lesion and then crossed to the right lesion via the connecting tunnel (Fig. IB). Venous return was documented through the right and left pulmonary veins. A gastrointestinal series gave no evidence of tracheoesophageal communication. The diagnosis of bilateral ILS was made, and bilateral resection (left lower lobectomy and right segmentectomy) was performed.
B Figure 1. Radiologie features of the bilateral intralobar sequestration (ILS) with a bridging isthmus. A. Chest computed tomography transverse section demonstrates multicystic lesions in the posterior, medial, and basal left and right lungs, which are connected by an air-filled bronchus (arrows). B. Three-dimensional reconstruction of the computed tomographic scan shows an aberrant artery (aal) branched from the left gastric artery via the celiac trunk and supplying the sequestration on the left. The aberrant artery is bifurcated immediately after insertion into the left ILS (aa2) and then crossed to the right ILS (aa3). Both lungs (right purple, left iight blue) are connected by a bronchus (light blue, arrow heads) derived from the left ILS.
He had no history of respiratory disease until this episode. Chest radiograph showed bilateral opacities occupying the left and right lower lobes. Computed tomography demonstrated a well-demarcated multicystic mass at the posteromedial bilateral lower lobe segments, which were connected by a bronchus behind the heart (Fig. lA). Contrast-enhanced computed tomography revealed an arterial supply from the left gastric artery via the celiac
56
R . FUKUZAWA ET AL.
METHODS AND PATHOLOGIC FINDINGS In order to examine the bronchial shape and course within the IES, we injected a radiopaque contrast medium into a bronchus in the isthmus before formalin fixafion. Both lungs contained sequestrated segments that were isolated from the normal bronchial tree but that could communicate by the connecting tunnel (Fig. 2B,C), confirming the diagnosis. The bronchi in the left sequestrated lung coursed laterally and upward from near the aberrant artery insertion site, and those in the right one ran from the bridging isthmus in a lateral direction (Fig. 2B,C). From the anomalous artery insertion site toward the hilus of the nonnal lung, the bronchi generally became smaller in diameter with increased branching in number and then formed acini (Fig. 2B,C). These features suggested that another bronchial system arose from near the anomalous artery insertion at the periphery of the lower lobe. Grossly, both lungs were connected by an isthmus covered by visceral pleura (Fig. 2A). Cut sections of both lungs showed a typical IES appearance: lesions consisting of lipid pneumonia with variably sized cystic changes were well-demarcated from the nonnal lung tissue. Histologically, the pulmonary parenchyma was distorted by chronic inflammation with lymphoid follicles. Dilated alveoli and bronchioles were filled with foamy macrophages. Both lesions contained congenital pulmonary airway malformation (CPAM) type II (Fig. 3A), but obstruction of a branch of the bronchus within the pulmonary parenchyma was identified only in the right sequestrated lung. The aberrant artery insertion site consisted of an elastic-type artery and bronchial tissue accompanied by cartilage, mucous glands, and smooth muscle, which looked like a pulmonary hilus (Fig. 3B). Stepwise serial sections taken from the communicating tunnel extending into the left sequestrated lung were all composed of similar bronchial tissue (Fig. 3C). These features were restricted to the region around the opening of the isthmus, and the normal segments of the left lower lobe demonstrated no significant pathologic change. DISCUSSION Since Pryce, who coined the term "sequestration," 1st hypothesized that traction by an anomalous branch of the aorta during segmentation of the developing lung resulted in separation from normal lung [7], a number of efiologic hypotheses have been proposed [2]. The hypothesis Gerle and colleagues [8] would seem to fit the findings in the
Figure 2. Pathologic features of tbe bilateral intralobar sequestration (ILS) witb a bridging isthmus. Gross feature of tbe bilateral ILS witb a bridging isthmus (arrow) (A). Radiographs showing tbe bronchial tree in botb tbe right (B) and left (C) sequestrated lungs. Note a catheter inserted into the aberrant artery near tbe stem bronchus-like structure. Both lungs contain tbe lesions isolated from tbe normal bronchial tree, confirming tbe diagnosis of ILS. Tbe shape and course of the bronchi demonstrate tbat tbe bronchial trees are not associated with the normal ones. Figure 3. Histologie features of the bilateral intralobar sequestration with a bridging isthmus. Congenital pulmonary airway malformation type II (A). Sections from near the aberrant artery insertion portion (B) and the bridging isthmus (C). The histologie features consist of bronchial tissue (br), cartilage (ca), gland (gl), and aberrant artery (aa), which resembles the pulmonary hilus. Hematoxylin and eosin, (A); elastic Van Gieson stain (B,C). Original magnification, x i o (A); x1.48 (B); X3.08 (C).
current case. They reported 3 cases of IES communicating with the gastrointestinal tract. They considered that ELS, IES, and sequestration that feature communication with the gastrointestinal tract share a common embryologie origin and suggested the term "congenital bronchopulmonary-foregut malfomiation" [8]. On histologie examination, our case showed that the region near the anomalous artery insertion site contained bronehial epithelial tissue with the presence of cartilage, smooth muscle, and bronchial glands, as well as elastic arteries branching from the anomalous artery (Fig. 3B,C). These structures resembled those making up the pulmonary hilus, and we therefore called them "pulmonary hilus-like structures." The following observations support this designation; (1) a bronchogram within the IES confirmed that the bronchi in both IESs were not derived from the normal bronchial tree, because the orientation of the bronchi was different from that of normal bronchi in the adjacent nornial lung (Fig. 2 B,C); (2) the aberrant elastic-type artery ran along the bronchi (Fig. 3B,C), which is characteristic of a pulmonary artery rather than a systemic artery. The question arises as to how such a hilus-like structure is formed. The anomalous artery has been
considered to be a remnant of a primitive aortic branch that originally had supplied the developing lung bud [9]. We speculate that persistence of the primitive aortic branch prevents regression of a pluripotent tissue, leading to development of an additional lung tissue that might eventually incorporate into the normal lung and form an ILS. In our case, we suggest that the lefr ILS initially developed from near the anomalous artery insertion portion where the hilus-like structure was histologically noted and that part of the lefr developing malformed lung tissue subsequently extended to the right side and gave rise to the bridging isthmus and the right IES. This pathogenetic assumption might be explained by the hypothesis that ILS originates from an aecessoty lung bud [8]. Recently, it has been thought that airway obstruction at bronchiolar formation levels during lung development (the psedoglandular phase: 7-17 weeks of gestation) consequently causes a parenchymal hamartomaous lesion referred to as CPAM [10]. In fact, Riedlinger and eollagues reported that bronchial atresia and CPAM were present in 82% and 91% of ILS, respectively [4]. Thus, the coexistence of bronchial atresia and CPAM in our BILATERAL ILS WITH BRIDGING ISTHMUS
57
case indicates that our bilateral ILS shares a common pathogenetic mechanism with conventional unilateral cases, supporting that ILS is a congenital anomaly. ACKNOWLEDGMENT This work was supported by Clinical Research Grants from the Tokyo Metropolitan Government and Grant-inAid for Scientific Research from the Ministry of Education, Culture, Sports, Science and Technology of Japan (25460427).
4.
5.
6.
7.
REFERENCES 1. Stocker JT, Maní H, Husain AN. The respiratory tract. In: Stocker JT, Dehner MD, Husain AN, eds. Stocker and Dehner's Pédiatrie Pathology. 3rd ed. Riverwoods, IL: Lippincott Williams & Wilkins, 2010;441-515. 2. Stocker JT, Malczak HT. A study of pulmonary ligament arteries: relationship to intralobar pulmonary sequestration. Chest 1984;86: 611-615. 3. Walford N, Htun K, Chen J, Liu YY, Teo H, Yeo GS. Intralobar sequestration of the lung is a congenital anomaly: anatomopatho-
58
R . FUKUZAWA ET AL.
8.
9.
10.
logical analysis of four cases diagnosed in fetal life. Pediatr Dev Pathol 2003;6:314-32I. Riedlinger WF, Vargas SO, Jennings RW, et al. Bronchial atresia is common to extralobar sequestration, intralobar sequestration, congenital cystic adenomatoid malfon^nation, and lobar emphysema. Pediatr Dev Pathol 2006;9:361-373. Kuo R, Shih SL, Huang JK. On the AJR digital viewbox: bilateral pulmonary sequestration with bridging isthmus in a boy with Williams syndrome. AJR Am J Roentgenol 2006;187:W133-W134. Cerruti MM, Marmolejos F, Caeciarelli T. Bilateral intralobar pulmonary sequestration with horseshoe lung. Ann Thorac Surg 1993;55:509-510. Pryce DM. Lower accessory pulmonary artery with intralobar sequestration of lung: a report of seven cases. J Pathol Bacteriol 1946;58:457^67. Gerle RD, Jaretzki A 3rd, Ashley CA, Berne AS. Congenital bronchopulmonary-foregut malformation: pulmonary sequestration communicating with the gastrointestinal tract. N Engl J Med 1968; 278:1413-1419. Ellis K. Fleischner lecture: developmental abnormalities in the systemic blood supply to the lungs. AJR Am J Roentgenol 1991; 156:669-679. Langston C. New concepts in the pathology of congenital lung malformations. Semin Pediatr Surg 2003;12:17-37.
Copyright of Pediatric & Developmental Pathology is the property of Allen Press Publishing Services Inc. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
