Management of antenatally diagnosed pulmonary sequestration associated with congenital cystic adenomatoid malformation

BACKGROUND: Sequestration with associated cystic adenomatoid malformation is rare. A study was undertaken to determine whether pulmonary sequestration associated with congenital cystic adenomatoid malformation has a more favourable natural history than that of sequestration without associated cystic adenomatoid malformation. METHODS: An outline of the postnatal work up leading to the management of extralobar or intralobar pulmonary sequestration with congenital cystic adenomatoid malformation diagnosed antenatally as pulmonary malformation is presented and the indications for surgical intervention are discussed. RESULTS: In five infants in whom an antenatal ultrasound scan had detected a congenital lung malformation at 18-19 weeks gestation a final diagnosis of extralobar or intralobar pulmonary sequestration with congenital cystic adenomatoid malformation was made postnatally. Postnatal ultrasound and computerised axial tomographic scans confirmed the diagnosis of sequestration by delineating anomalous vascular supply. Cystic changes were also observed in the basal area of the sequestration in all patients. Four children remained asymptomatic and one infant presented at 10 months of age with pneumonia. The mean age at surgical resection was 6.8 months (range 2-10). Histopathological examination confirmed intralobar pulmonary sequestration with associated Stocker type 2 congenital cystic adenomatoid malformation in two patients and extralobar pulmonary sequestration with associated Stocker type 2 congenital cystic adenomatoid malformation in three patients. The mean period of follow up was four years (range 1-8). The children remain well and are developing normally. CONCLUSIONS: The importance of seeking an anomalous blood supply in children with congenital lung lesions is emphasised. Pulmonary sequestration and congenital cystic adenomatoid malformation probably share a common embryogenesis despite diverse morphology. The natural history of antenatally diagnosed lung masses is variable. Early postnatal surgical resection of pulmonary sequestration with cystic adenomatoid malformation is recommended. Surgical excision should be conservative, sparing the normal lung parenchyma.     

Experience with thoracoscopic lobectomy in infants and children

Purpose: This study evaluates the safety and efficacy of thoracoscopic lobectomy in infants and children. Methods: From January 1995 to May 2002, 45 patients underwent video-assisted thoracoscopic lobe resection. Ages ranged from 2 days to 18 years and weights from 2.8 to 78 kg. Preoperative diagnosis included sequestration/congenital adenomatoid malformation (n = 28), severe bronchiectasis (n = 12), congenital lobar emphysema (n = 3), and malignancy (n = 2). Results: Forty-three of 45 procedures were completed thoracoscopically. Operating times ranged from 35 minutes to 210 minutes (average, 125 minutes). There were 6 upper, one middle, and 38 lower lobe resections. There was one intraoperative complication (2.4%) requiring conversion to an open thoracotomy. Chest tubes were left in 38 of 45 patients for 1 to 3 days. Hospital stay ranged from 1 to 5 days (average 2.4). Conclusions: Thoracoscopic lung resection is a safe and efficacious technique. It helps avoid the inherent morbidity of a major thoracotomy incision and is associated with the same decrease in postoperative pain, recovery, and hospital stay as seen in minimally invasive procedures. J Pediatr Surg 38:102-104.     

Experience with 144 consecutive pediatric thoracoscopic lobectomies

OBJECTIVE: The early experience with the technique and short-term outcomes after pediatric thoracoscopic lobectomy were independently reported by the authors several years ago. This paper updates their combined experience, evaluating the safety, efficacy, and long-term outcomes. METHODS: From January 1995 to May 2005, 144 consecutive patients underwent a thoracoscopic lobectomy. Preoperative diagnoses included cystic adenomatoid malformation/sequestration (n = 112), bronchiectasis (n = 19), lobar emphysema (n = 10), and malignancy (n = 3). Ages ranged from 2 days to 18 years, and weight ranged from 2.8 to 78 kgs. Three or four valved ports were used with a controlled pneumothorax. Single-lung ventilation was used in all cases. Follow-up ranged from 1 to 10 years. RESULTS: All but three procedures were completed thoracoscopically; one was converted to repair an injured upper lobe bronchus during a lower lobectomy, one resulting from bleeding, and another caused by what was believed to be a potentially inadequate margin during the resection of a large tumor. The operating time ranged from 35 to 220 minutes (median, 125). There were 110 lower, 24 upper, and 10 middle lobe resections. There was one intraoperative complication--the compromise of a left upper lobe bronchus. There were four postoperative complications: pneumonia, pneumothorax, empyema, and prolonged chest tube drainage. There were no reoperations. The median duration of hospital stay was 2.8 days. A long-term follow-up revealed no cases of musculoskeletal deformity or weakness. CONCLUSIONS: The current techniques and equipment allow for the complete thoracoscopic resection of pulmonary lobes in any age or size of a child, with low morbidity and no mortality. Long-term outcomes support the efficacy of this technique that spares growing children a thoracotomy that has the potential for late musculoskeletal morbidity.     

Congenital cystic adenomatoid malformation detected by prenatal sonography; report of 2 cases

We report 2 cases of congenital cystic adenomatoid malformation (CCAM) detected by prenatal sonography. The first CCAM was diagnosed by fetal sonography in a female fetus at 30 weeks' gestation. The infant was born at 37 weeks' gestation, with a body weight of 2,770 g. After birth, chest computed tomography (CT) showed a multicystic mass in the middle lobe of the lung. She remained asymptomatic until age 21 months, when she suffered pneumonia. Two months later, middle lobectomy was performed. The second CCAM was diagnosed by fetal sonography in a female fetus at 25 weeks' gestation. She was born at 39 weeks' gestation, with a body weight of 3,292 g. Four days after birth, CCAM type II was diagnosed by chest CT. The infant was asymptomatic, and left lower lobectomy was performed 11 months after birth.     

The changing spectrum of cystic pulmonary lesions requiring surgical resection in infants

Congenital labor emphysema and cystic adenomatoid malformation have been the most common surgically treatable cystic lesions of the lung. With the successful treatment of newborn respiratory distress syndrome, an increased frequency of an acquired form of cystic disease, interstitial pulmonary emphysema, has been observed. Some degree of interstitial pulmonary emphysema is relatively common, and during the years 1980 to 1983 this disease was diagnosed in 372 infants. Seven of these infants, all premature, underwent resection of relatively localized areas of persistent cystic interstitial pulmonary emphysema. Lobectomy or wedge resection was performed in five patients because of their inability to be weaned from the ventilator. A sixth patient with this disease underwent lobectomy for recurrent pneumothoraces. A seventh patient underwent lobectomy because the cystic interstitial pulmonary emphysema produced atelectasis and recurrent infections. All seven patients were extubated by the fourth postoperative day, have been discharged, and are showing respiratory improvement. Within the same period, four infants had congenital lobar emphysema and two had congenital cystic adenomatoid malformation. They were gestational age 36 weeks or older and, although respiratory distress was present to some degree in all six, only one was ventilator dependent at operation. All underwent lobectomy and one infant had a left upper lobe resection and right middle lobectomy on separate occasions for bilateral congenital cystic adenomatoid malformation. All patients with congenital labor emphysema and congenital cystic adenomatoid malformation have been discharged and are doing well. Our results suggest the following conclusions: Persistent interstitial pulmonary emphysema is now the most common indication for pulmonary resection in the newborn period. The anatomic distribution of resected interstitial pulmonary emphysema is similar to that of congenital lobar emphysema. Although only a small fraction (less than 2%) of patients with interstitial pulmonary emphysema require operation, the development of relatively large discrete cystic areas that (1) significantly decrease effective lung volume and produce respirator dependence, (2) produce atelectasis and recurrent infections, or (3) lead to pneumothoraces may make pulmonary resection beneficial. Finally, despite the presence of generalized lung disease in patients with interstitial pulmonary emphysema, these patients can be expected to improve significantly after resection, and the long-term outcome is generally good.     

Video-assisted thoracoscopic lobectomy in the treatment of intralobar pulmonary sequestration

Pulmonary sequestration is a congenital malformation characterised by cystic, non-functioning embryonic lung tissue with vascularisation of an abnormal systemic artery. They are classified as intralobar (75%) and extralobar (25%) and are more common in the left lung and lower lobes (60-90%). We report two cases of intralobar pulmonary sequestration located in the lower lobe of the left lung which were subjected to video-assisted thoracoscopic surgery (VATS). Both patients had recurrent infections for which, after performing imaging tests, they were diagnosed with intralobar pulmonary sequestration in the left lower lobe, with an afferent arterial branch to the malformation from the aorta. A lower lobectomy was performed by video-assisted surgery, dividing the aberrant aortic artery with an endostapler. A single thoracic chest tube was placed and removed on postoperative day 2 and the patients were discharged on the same day. In both cases, the pathology examination revealed intralobar pulmonary sequestration. Pulmonary sequestrations are uncommon malformations that can be operated on using minimally invasive techniques, thereby permitting early discharge and a low rate of complications.     

Clinical characteristics and feasibility of thoracoscopic approach for congenital cystic adenomatoid malformation in adults.

OBJECTIVE: Congenital cystic adenomatoid malformation (CCAM) in adults is very rare, and surgical resection is the treatment of choice. We evaluated the clinical characteristics and feasibility and safety of video-assisted thoracic surgery (VATS) in adult patients with CCAM. METHODS: Retrospective analysis of nine consecutive adult patients with CCAM treated surgically between January 1995 and April 2006 at a single center. The clinical characteristics and surgical outcome were evaluated retrospectively. RESULTS: The records of five men and four women were examined. The median age at diagnosis was 30.5 years (range 16-44 years). The most frequent symptoms were cough (n=4) and hemoptysis (n=2). Three patients had recurrent respiratory infections. Chest computed tomography (CT) was reviewed in all cases. The lesions were on the right side in six patients and on the left in three. All patients had lesions in a single lobe, except one patient who had multi-lobar lesions. The CCAM appeared as thin-walled multi-septated cystic lesions in six patients and as parenchymal consolidations in three patients. Four patients underwent VATS lobectomy and four underwent lobectomy through a conventional open thoracotomy. One patient had open pneumonectomy. The median operating time (160 min in VATS lobectomy and 165 min in open lobectomy), median duration of chest tube drainage (4 days in VATS lobectomy and 6.5 days in open lobectomy), and postoperative complications (one patient in each treatment group) were similar between the VATS and open lobectomy groups. The hospital stay was shorter in the VATS lobectomy group (median 5 days) than in the open lobectomy group (median 7.5 days). No postoperative or procedure-related mortality occurred. CONCLUSIONS: CCAM is rare in adults. Patients usually present with recurrent respiratory infections and thin-walled multi-septated cystic lesions. VATS is a feasible and safe technique to treat adult patients with CCAM.     

Congenital bronchopulmonary malformations. Diagnostic and therapeutic considerations

Congenital bronchopulmonary malformations are uncommon but potentially life-threatening anomalies of infants and children. Between 1970 and 1988, 45 patients from birth to 13 years of age (23 boys and 22 girls) underwent evaluation and treatment for bronchopulmonary malformations. Thirty-seven had solitary lesions: bronchogenic cyst (n = 13), cystic adenomatoid malformation (n = 9), congenital lobar emphysema (n = 6), pulmonary sequestration (n = 6), arteriovenous malformation (n = 2), and bronchial atresia (n = 1). Eight additional patients had two simultaneous abnormalities and three patients had congenital diaphragmatic hernias. Twenty-one patients had respiratory symptoms, which were severe in seven. Twelve had pulmonary infection and 10 patients were completely free of symptoms. Plain chest roentgenogram was the only diagnostic imaging performed in 11 patients. Thirteen patients underwent computed tomographic scan, but in only four was it essential for diagnosis. Prenatal ultrasonography in three patients demonstrated cystic adenomatoid malformation in two, with one false negative study. Postnatally, ultrasonography was also useful in establishing the diagnoses of cystic adenomatoid malformation and pulmonary sequestration. Thoracotomy with excision of the lesion by lobectomy or pneumonectomy resulted in survival of 42 patients (93%). Three deaths in neonates were due to pulmonary hypoplasia and hypertension. Two of them had concomitant diaphragmatic hernia; the other had a cystic adenomatoid malformation and died despite the use of postoperative extracorporeal membrane oxygenation. These data demonstrate that congenital bronchopulmonary malformations usually can be diagnosed by plain chest x-ray films. Ancillary studies such as ultrasonography or computed tomography may occasionally be necessary. Combinations of the different types of bronchopulmonary malformations occurred frequently. All lesions, including symptomatic lesions in neonates, can be managed surgically soon after diagnosis.     

First decade's experience with thoracoscopic lobectomy in infants and children

Purpose

This study evaluates the safety and efficacy of thoracoscopic lobectomy in infants and children.

Methods

From January 1995 to March 2007, 97 patients underwent video-assisted thoracoscopic lobe resection. Ages ranged from 2 days to 18 years and weights from 2.8 to 78 kg. Preoperative diagnosis included sequestration/congenital adenomatoid malformation (65), severe bronchiectasis (21), congenital lobar emphysema (9), and malignancy (2).

Results

Of 97 procedures, 93 were completed thoracoscopically. Operative times ranged from 35 minutes to 210 minutes (average, 115 minutes). There were 19 upper, 11 middle, and 67 lower lobe resections. There were 3 intraoperative complications (3.1%) requiring conversion to an open thoracotomy. Chest tubes were left in 88 of 97 procedures for 1 to 3 days (average, 2.1 days). Hospital stay ranged from 1 to 12 days (average, 2.4 days).

Conclusions

Thoracoscopic lung resection is a safe and efficacious technique. It avoids the inherent morbidity of a major thoracotomy incision and is associated with the same decrease in postoperative pain, recovery, and hospital stay as seen in minimally invasive procedures.     

Congenital lung malformations--antenatal and postnatal evaluation and management.

We reviewed our institutional experience with pulmonary resection for congenital bronchopulmonary malformations and analysed the management and outcome of pregnancies with a prenatal diagnosis of congenital lung malformations. Between January 1993 and December 2003, 31 patients underwent evaluation and pulmonary resection for bronchopulmonary malformations. Common clinical presentations were respiratory distress (9), respiratory infections/pneumonias (22), and dyspnoea (9). Diagnostic modalities included chest radiography, CT scan (22), MRI scan (7), arteriography (1), and bronchoscopy (5). There were 13 congenital cystic adenomatoid malformations (CCAM), six pulmonary sequestrations, three bronchogenic cysts, and nine congenital lobar emphysemas (CLE). Fifteen patients who underwent resection were diagnosed by antenatal ultrasound. No foetus had hydrops or associated malformations. No pregnancy was terminated. There was no foetal demise. Regression of the sonographic appearance was observed in six cases. Amniotic puncture was required for hydramnios in three cases. Eight emergency resections were performed (CCAM 4; CLE 3; Bronchogenic cyst 1). Surgical procedures included 24 lobectomies, one right middle lobectomy with a wedge resection of the right lower lobe, one completion right lower lobectomy, four sequestrectomies, one mediastinal mass excision and one wedge resection for a bronchogenic cyst. There were no deaths. Postoperative complications included: persistent air leak (n=2; one requiring completion lobectomy) and pneumothorax (1). Persistent mild symptoms were present in five patients, at long-term follow-up. Congenital cystic adenomatoid malformation and congenital lobar emphysema were the commonest congenital anomalies. Congenital lung malformations are increasingly diagnosed antenatally, sometimes necessitating emergent surgical resection. The natural history is variable. All infants with a prenatal diagnosis require postnatal evaluation. Patients should be evaluated for associated disorders. The presence of mass effects is an indication for therapeutic decompression. The risk of pulmonary compression, infection and malignant degeneration makes resection imperative, even in asymptomatic patients. Lobectomy is the procedure of choice, is well tolerated, and leads to excellent outcomes.     

Infected cystic adenomatoid malformation in an adult

Congenital cystic adenomatoid malformation of the lung is rare in adults. We present a 51-year-old man with recurrent lower respiratory tract infections and intermittent febrile episodes for more than 10 years. Multiple cavitary lesions with fluid accumulation were seen in the right lower lobe of the lung on radiography and computed tomography of the chest. Wedge resection of the right lower lobe of the lung was performed through a mini-thoracotomy using video-assisted thoracoscopy. Multiple infected cysts were found in a 10 × 8 × 6 cm area of lung. The histologic diagnosis was infected type I congenital cystic adenomatoid malformation. The patient remains well 1 year after the operation.     

Complex extralobar sequestration in a 24-year-old woman

Extralobar sequestration is a congenital acquired disease more frequently observed after birth or during infancy. In half of the cases, it is associated with another malformation. In the following case we report the observation of a 24-year-old female with right extralobar sequestration associated with a diaphragmatic hernia and containing gastric mucosa and a congenital cystic adenomatoid lung malformation. Such malformative lung tissue is known to potentially degenerate and justifies surgery even when patients are asymptomatic.     

Congenital Parenchymatous Malformations of the Lung

Congenital lung malformations, primary and secondary, contribute to an important portion of pediatric thoracic surgery. One purpose of this report is to outline the close relation in terms of embryology and clinical presentation of congenital parenchymatous pulmonary malformations. In a retrospective study we also aim to evaluate our experience with the diagnosis and surgical management of congenital parenchymatous bronchopulmonary malformations and to compare our data with the literature. From January 1979 to December 1996 a series of 48 patients, 30 males (62.5%) and 18 females (37.5%), were operated on for congenital bronchopulmonary malformations. Pulmonary sequestration, bronchogenic cysts, congenital lobar emphysema, and congenital cystic adenomatoid malformation were seen in 16, 13, 5, and 14 patients, respectively. The first clinical symptoms occurred at a mean age of 8.8 years (1 day to 62 years), and the mean age at the time of surgical intervention was 9.3 years (1 day to 62 years). The maximum time between first symptoms and surgical treatment was 27 years. A lobectomy was performed in 22 cases; in the other patients more lung-preserving surgery such as enucleation or sequestrectomy was performed. Only one postoperative death occurred following lobectomy for pulmonary sequestration, and it was due to pulmonary hypoplasia and pulmonary hypertension. Eleven other patients presented with postsurgical complications: pneumothorax (n= 5), pleural effusion (n= 3), prolonged air leak (n= 2), portal vein thrombosis (n= 1), and hemorrhage requiring reintervention (n= 1). We conclude that any thoracic cystic lesion expanding on chest radiography should be an indication for surgical resection, even if asymptomatic, because of the risk of pulmonary compression, infection, or malignant degeneration. In the few cases of a fetal intrathoracic mass, prenatal diagnosis and intrauterine intervention may be indicated, and these indications are also discussed.     

Congenital cystic adenomatoid malformation of the lung

Twenty cases of cystic adenomatoid malformation of the lung were observed: 2 had died in utero; the diagnosis was made at birth in 13 infants of which 3 were premature. A Bochdalek's hernia had been diagnosed before birth in 2 cases by echo-tomography. The correct antenatal diagnosis had been made in 2 cases. Two infants had no symptoms, 3 were dyspneic, 8 were in respiratory distress and had to be intubated and ventilated. Two had a prune belly syndrome. Eight infants had a thoracotomy during their first week of life. Pulmonary resections concerning those 13 patients comprised 10 lower lobectomies, 1 of which was associated with a lingulectomy, and 2 upper lobectomies, 1 of which was associated with a middle lobe resection. Five patients were diagnosed and operated upon between 10 months and 8 years of age; 4 had recurrent bronchitis and 1 was diagnosed during the treatment of a gastroenteritis. They had 3 lower and 2 upper lobectomies. Recovery was uneventful in all patients except for 1 who was reoperated upon for intestinal obstruction. Antenatal diagnosis of cystic adenomatoid malformation should become standard. The malformation may be mistaken for a pulmonary sequestration or bronchogenic cyst. Differential diagnosis of a congenital hernia is important.     

Treatment of pulmonary sequestration by video-assisted thoracic surgery (VATS)

Pulmonary sequestration is a rare malformation of the respiratory tract that accounts for 0.15–6.4% of all congenital lung anomalies. Treatment requires resection of the lesion, provided that there is no technical contraindication. The lession should first be evaluated using video thoracoscopy and then resected whenever possible by video-assisted thoracic surgery (VATS). We report a case of extralobar pulmonary sequestration in a 48-year-old woman. She underwent lobectomy by VATS and achieved an excellent outcome.     

Video-assisted thoracic surgery for bronchopulmonary sequestration

Bronchopulmonary sequestration is a rare congenital lung anomaly for which surgical resection is the definitive treatment. Open thoracotomy is the conventional approach, yet associated with considerable morbidity. We report one of the largest series of major lung resection for bronchopulmonary sequestration using the video-assisted thoracic surgery (VATS) approach that could reduce such morbidity. Six cases of VATS anatomical lobectomy for intrapulmonary sequestration performed between January 1996 and January 2005 were reviewed. The six patients included two males and four females, with a mean age of 43.3 years (range: 27-64 years). Anatomical lobectomy without conversion to open was achieved in all cases. The mean operating time was 112.8 min (range: 90-140 min), the mean blood loss was 283.3 ml (range: 100-500 ml), and the mean length of post-operative hospital stay was 8.8 days (range: 7-24 days). There was no mortality. Three patients had minor wound infection. The results were comparable patients receiving lung resections for bronchopulmonary sequestration by an open approach. VATS major lung resection for bronchopulmonary sequestration is safe and feasible. Further studies are warranted to define the role of VATS in the management of bronchopulmonary sequestration.     

Diagnosis and management of congenital cystic disease of the lung in children

Pulmonary sequestration, congenital adenomatoid malformation (CAM), congenital lobar overinflation (CLO), and bronchogenic cysts are four congenital lesions that may present as abnormal cystic areas within the pleural cavity in early life. They share similar clinical and embryologic characteristics, are frequently difficult to diagnose, and all require surgical treatment. From December 1974 to January 1985, 22 patients were operated upon on the Pediatric Surgical Service for congenital cystic disease of the lung. There were eight females and 14 males, ranging in age from 1 day to 18 years; 12 were under 6 months of age, and 14 were under one year. There were 7 CAMs, 5 extralobar pulmonary sequestrations, 5 intralobar pulmonary sequestrations, 3 bronchogenic cysts, and 3 cystic lobes caused by CLO. One patient had bilateral pulmonary sequestrations, intralobar on one side and extralobar on the other, with both sharing a common systemic artery arising from the infradiaphragmatic aorta. All except two asymptomatic patients presented with either progressive respiratory distress or recurrent pulmonary infections, and had cystic changes noted on chest roentgenograms. The differentiation of these congenital lesions from resolvable inflammatory cysts is important. Diagnosis was aided by selective utilization of barium contrast studies, ultrasonography, computed tomography, and arteriography. Operation consisted of resection alone of the extralobar pulmonary sequestrations and bronchogenic cysts, and total lobectomy in CAM, intralobar sequestration, and CLO. One infant with CAM died 1 day postoperatively from bilateral hypoplastic lungs. The other 21 patients are alive and well with follow-up ranging from 1 month to 9 years, with a mean of 3.5 years.(ABSTRACT TRUNCATED AT 250 WORDS)     

Intrapulmonary sequestration operated by video-assisted thoracoscopic surgery lobectomy; report of a case

Pulmonary sequestration is a rare malformation of the respiratory tract. We here report an adult case of intralobar pulmonary sequestration with aberrant artery of the right lower lobe. A 20-year-old man admitted to our hospital with a chest discomfort. Chest computed tomography (CT), angiography, and scintigraphy showed abnormal findings of the right lower lobe with an aberrant artery. Under the diagnosis of pulmonary sequestration, video-assisted thoracoscopic surgery (VATS) lobectomy was performed. VATS is useful for the operation of pulmonary sequestration.     

Evolving experience with video-assisted thoracic surgery in congenital cystic lung lesions in a British pediatric center

Background/Purpose

Video-assisted thoracic surgery (VATS) is increasingly used for the resection of congenital cystic lung lesions (CLLs). This study aimed to evaluate the efficacy of VATS and its outcome in both antenatally and postnatally detected CLLs.

Methods

Forty-six patients managed during 2000-2005 were studied. Demographics, investigations, operative details, and outcome data were collected and evaluated. Patients were divided into 3 groups for analysis.

Results

Antenatally diagnosed (groups I and II, n = 35): group I (20) had VATS at 20 months median (range, 16-35 months). Video-assisted thoracic surgery was successful in 14 of 20 (70%), notably in all cases of extralobar sequestrations and foregut duplication cysts. Inadequate vision/lung collapse and technical difficulties were the main reasons for conversion to open thoracotomy. Group II (n = 15) was considered unsuitable for VATS because of neonatal symptoms (6 congenital cystic adenomatoid malformations of the lung [CCAMs]) and/or large size/inexperience (5 CCAMs, 4 sequestrations) and had elective thoracotomy at 8 months median (range, 6 days-20 months).Postnatally diagnosed (group III, n = 11): 3 CCAMs, 6 duplications, and 2 sequestrations were diagnosed because of recurrent chest infection (8) or stridor (2), or incidentally (1) at 8 years median (range, 1.2-14 years). Video-assisted thoracic surgery was successful in 3 foregut duplications. A duplication and an intralobar sequestration were converted; open thoracotomy was performed in others because of previous recurrent pneumonic episodes.Postoperative pain and hospital stay were significantly less (P < .001) in successful VATS resection: median of 2 days (range, 1-7 days) compared with thoracotomy median of 6 days (range, 4-20 days).

Conclusions

Video-assisted thoracic surgery is a safe and effective option for asymptomatic congenital CLLs. It is anticipated that more successful CCAM resections using VATS will occur in the future as our technical ability improves.     

Congenital cystic adenomatoid malformation connected to an extralobar pulmonary sequestration in the contralateral chest: common origin?

Extralobar pulmonary sequestration (ELS) and congenital cystic adenomatoid malformation have been reported to coexist in several variations. This suggests a common embryologic origin. A 6-month-old boy presented with a history of recurrent pneumonias. The patient was diagnosed with a right lower lobe congenital cystic adenomatoid malformation (CCAM) and a left lower lobe ELS/CCAM. The diagnosis was made with the aid of a multidetector computed tomography (CT). Three-dimensional CT reconstruction showed the presence of a right lower lobe CCAM, a left lower ELS with an aberrant arterial supply from the celiac axis, and possible venous drainage into the right CCAM. The patient underwent a right thoracotomy. Intraoperatively, the lesions were discovered to be connected by a band of tissue. The right lower lobe CCAM and the left ELS were removed from the right chest. Histologic analysis confirmed the presence of a CCAM within the right lower lobe. The ELS had involvement of a type II CCAM within the sequestration. The connection between the right CCAM and left ELS/CCAM showed an anomalous conducting airway, anomalous vein, and anomalous artery connecting the 2 lesions. The authors present the first case of a CCAM connected to an ELS/CCAM in the contralateral hemithorax. The unique anatomic configuration of these lesions suggests a common embryologic origin of ELS and CCAM.