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J Thorac Cardiovasc Surg 2003;126:574-581
© 2003 The American Association for Thoracic Surgery

General thoracic surgery

Pneumonectomy in children for destroyed lung and the long-term consequences

evval Eren, MD^a,*, Mehmet Nesimi Eren, MD^a, Akin Eraslan Balc, MD^a

^a Department of Thoracic and Cardiovascular Surgery, Dicle University School of Medicine, Diyarbakir, Turkey

Read at the annual meeting of the European Society of Thoracic Surgeons, Istanbul, Turkey, Oct 26-28, 2002.

Received for publication December 10, 2002; revisions received February 7, 2003; revisions received February 24, 2003; accepted for publication March 18, 2003.

^* Address for reprints: evval Eren, MD, Akkoyunlu 3.sok. Altunbay 3 Apt. No. 7, 21100, Diyarbakir, Turkey
sevval{at}dicle.edu.tr

	Abstract

Top Abstract Materials and methods Results Discussion Conclusion References

 
OBJECTIVES: Destroyed lung introduces irreversible changes in lung parenchyma. This condition is uncommon in children. Operative intervention is essential for children in this state. We demonstrate our experience with this condition and report on the respective long-term results.

METHOD: Seventeen children who underwent pneumonectomy for destroyed lung during a 15-year period were retrospectively analyzed. Long-term results were detected in 13 patients.

RESULTS: Seventeen children underwent pneumonectomy. Five children were female (29.4%), and 12 children were male (70.5%). The median age of the study group children was 9.1 years (3-16 years). Sputum was the most common presenting symptom (n = 13, 76.4%). Bronchiectasis (n = 11), tuberculosis (n = 4), and necrotizing lung disease (n = 2) were the main underlying conditions. Destroyed lung was detected on the left side in 14 children (82.4%) and on the right side in 3 children (17.6%). Main bronchial stenosis was found in 4 children and mucosal thickening or congestion in 5 children. The median length of hospital stay was 15.5 days. The mortality rate was 11.7% (n = 2), and the morbidity rate was 23.5% (n = 4). Follow-up information was available for 13 patients, and this ranged from 1 year to 12 years (median 5.2 years). The respiratory capacity and scoliosis level of the patients were examined.

CONCLUSIONS: Although pneumonectomy is considered a difficult procedure in children, its use for destroyed lung resolves complications and improves a patient’s quality of life. In time, the remaining lung expands to compensate for the loss of the removed lung. Children grew and developed normally after pneumonectomy. Patients tend not to have major skeletal deformation as the result of pneumonectomy in the short term.

Key Words: 11

Destroyed lung is an uncommon condition in children causing irreversible changes in lung parenchyma, and surgical intervention becomes essential. Destroyed lung is most often caused by inflammatory lung diseases such as tuberculosis, whole lung bronchiectasis, necrotizing pneumonia, multiple or extensive lung abscesses, fungal infections, lung gangrene, and mycobacteria other than tuberculosis.^1-7 Other important causes include bronchial stricture and congenital malformations.^1,2

Destroyed lung gives rise to chronically morbid and sometimes acute life-threatening complications such as massive hemoptysis, empyema, secondary fungal infections, secondary amyloidosis, septicemia, and pulmonary-systemic shunting.^2,5,8,9 Surgical resection in destroyed lung is used to resolve complications and improve a patient’s quality of life.⁵ Almost all of our patients who underwent pneumonectomy had undergone numerous treatments. However, irregular and inadequate treatment, the cessation of medication shortly after symptom improvement, and a lack of check-ups after treatment are among the factors that accelerate the need to perform pneumonectomy in children.

The long-term outlook for the respiratory function of children after pneumonectomy is good. Children are able to perform daily activities and exercises without difficulties despite reduced pulmonary reserves.¹⁰

This retrospective study was undertaken to review our clinical experience and long-term results for pneumonectomy in children with destroyed lung.

	Materials and methods

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The files of all patients who underwent pneumonectomy at Dicle University School of Medicine, Department of Thoracic and Cardiovascular Surgery, between 1987 and 2002 were reviewed. Patient demographics, medical history, presenting symptoms, causative factors, preoperative evaluations, treatment, operative procedures, postoperative course, and pathologic findings were reviewed, and the follow-up results were evaluated.

	Results

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History and symptoms
Seventeen patients, 12 boys and 5 girls, with an average age of 9.1 years (range 3-16 years), underwent pneumonectomy. The most common underlying disease was bronchiectasis (n = 11); other underlying diseases included tuberculosis (n = 4) and necrotizing pneumonia (n = 2). In 2 patients with bronchiectasis, the diagnosis was congenital cystic bronchiectasis. All patients except 2 were hospitalized for treatment (Table 1). Two patients who had received treatment for 1 year or less demonstrated necrotizing pneumonia. One patient with tuberculosis did not receive any antituberculosis treatment during the preoperative period, whereas 2 patients received irregular or inadequate treatment. The diagnosis of the patient with tuberculosis who did not receive prior treatment was made by postoperative histopathologic examination. We did not use routine perioperative antituberculous chemotherapy unless there was evidence of active disease.

Radiologic and diagnostic examinations
Destroyed lungs were present on the left side in 14 patients (82.3%) and on the right side in 3 patients (17.6%). Radiologic diagnostic methods included chest radiography in all patients, chest computed tomography (CT) in 14 patients (Figure 1), bronchography in 8 patients (Figure 2), and pulmonary ventilation-perfusion scan in 4 patients. Main bronchial stenosis was found in 4 patients and mucosal thickening and congestion in 5 patients by means of bronchoscopy or bronchography (Figure 2).

View larger version (100K): [in this window] [in a new window]   Figure 1. Chest CT scan of a 4-year-old boy shows a destroyed left lung.

View larger version (97K): [in this window] [in a new window]   Figure 2. Bronchogram of a destroyed left lung shows stenosis of the left main bronchus.

It was possible to perform pulmonary function tests in 8 of the older children and in those able to cooperate. The other 9 younger patients were evaluated by an exercise-tolerance test. The 6-minute walk test was performed along a level hospital corridor. Oxygen saturation (SaO₂) was measured by finger pulse oximetry before and during the test and monitored continuously. Measurements were recorded at 30-second intervals. All patients were able to walk continuously for the 6-minute period. The lowest SaO₂ was used to calculate minimum SaO₂. Mean minimum SaO₂ was 95.3. Care was taken to ensure that SaO₂ did not decrease less than 90%. Along with exercise, a 2% or more desaturation was considered to represent a risk. One patient seen to be at risk received 3 weeks of additional chest physiotherapy, incentive spirometry, nutritional support, and ambulation with physical therapy. The patient underwent operation after this course of treatment. No patient was observed to have effort dyspnea after the test. In addition, blood samples were taken from all patients for blood gas analysis from the femoral artery.

According to the nutritional status (size and weight) of the children preoperatively, 3 children were in the 3rd to 10th percentile, 9 children were in the 10th to 25th percentile, and 2 children were in the 25th to 50th percentile.

Operative course
All of the operations were elective. Fourteen patients (82.3%) underwent left pneumonectomy, and 3 patients (17.6%) underwent right pneumonectomy. A double-lumen endotracheal tube was used in older children (n = 7) to avoid the spillage of infected material into the contralateral bronchus. In most of the patients in which the double-lumen endotracheal tube was not used, bronchoscopy was performed, and the bronchus of the side ready for resection was cleaned by aspiration before the introduction of an endotracheal tube. Frequent intraoperative aspiration was required for these patients. A Fogarty embolectomy catheter was used as a bronchus blocker in 2 patients, with unsatisfactory results. In these 2 patients, respiratory distress occurred because the Fogarty catheter was either not properly placed or the patient was moved and the catheter was disturbed. As a result of this, the Fogarty catheter was withdrawn, and the surgery was continued. The standard posterolateral thoracotomy approach was used in all patients. Thoracotomy was performed in a way to conserve as much muscle as possible. All of the pneumonectomies were performed in the intrapleural plane.

Intrapericardial pneumonectomy was performed in 2 patients. The main bronchus was closed by stapler in 2 patients and by hand suturing with nonabsorbable suture material (polypropylene) or absorbable Vicryl polyglactin 910 (Ethicon, Inc, Somerville, NJ) in the other patients. The bronchial stump was routinely covered by adjacent tissue, mediastinal pleura, pericardium, intercostal muscle, or a combination of these. Ten patients underwent invasive blood pressure monitoring, which also allowed regular blood gas analysis during the intraoperative and postoperative period. One intraoperative death occurred as the result of respiratory failure and cardiac arrest. All patients except one were extubated in the operating theater. One patient remained in the recovery room awaiting extubation for 6 hours because of low oxygen saturation levels. A chest tube was routinely placed into the pleural cavity and removed on the first or second postoperative day in most patients.

Postoperative course and complications
Postoperative complications were followed for 30 days. The postoperative morbidity rate was 23.5% (n = 4). One patient developed empyema (left) without bronchopleural fistula (BPF) on postoperative day 7. This patient was treated by closed tube thoracostomy, antibiotics according to a culture antibiogram, and postpneumonectomy space irrigation with a 5% dextrose solution containing 2 g of cephalosporin through a catheter introduced from the midclavicular line at the second intercostal space. A continuous inflow-outflow irrigation system was established through the pleural cavity at an infusion rate of 50 mL per hour. After 2 weeks, the pleural fluid color had cleared. After cultures proved negative for 3 days, tube drainage was discontinued and pleural fluid was allowed to reaccumulate to fill the remaining space. Postoperative hemorrhage occurred from the intercostal muscle in 1 patient, and this required rethoracotomy. Respiratory inadequacy occurred in 1 patient as a result of atelectasis, which was treated by bronchoscopic aspiration and respiration exercise. Empyema and BPF developed on postoperative day 25 in 1 patient; open-window dressing was performed in this patient after closed thorax drainage. Later, BPF closure and myoplasty procedures were performed to obliterate the postpneumonectomy space. This patient underwent a right pneumonectomy and had a history of tuberculosis. There was 1 postoperative death after a left pneumonectomy as the result of postpneumonectomy edema on postoperative day 3. The median length of inpatient hospital stay was 15.5 days. Diagnosis was made by histopathologic examination of the resected lung tissue in all patients. The pathologic findings in the resected pneumonectomy specimens were end-stage destroyed lung.

Follow-up
Follow-up information was available for 13 patients, which ranged from 1 to 12 years (median 5.2 years). One patient died 2 years after surgery because of infections in the remaining lung. This patient underwent myoplasty as the result of BPF and empyema. Two children had respiratory infections that were managed by outpatient therapy. The other patients had no associated problems. There was marked herniation of the remaining lung with a mediastinal shift to the opposite side on follow-up chest radiography or CT of all the patients (Figure 3).

View larger version (95K): [in this window] [in a new window]   Figure 3. Chest CT scan of an 8-year-old boy shows the right lung goes into the left chest with displacement of the mediastinal structures into the left hemithorax 4 years after pneumonectomy.

View larger version (64K): [in this window] [in a new window]   Figure 4. A, Chest x-ray film of a 7-year-old boy shows a light degree of scoliosis with collapse of the left intercostal space 3 years after pneumonectomy. B, Chest x-ray film of an 18-year-old man shows a light degree of scoliosis with collapse of the left intercostal spaces 12 years after pneumonectomy.

In addition, we evaluated all tests assessing the restriction or obstruction of respiratory functions. According to VC, 7 patients had light restrictions (VC: 66%-80% predicted), and 6 patients were shown to have medium restrictive ventilation dysfunction (VC: 51%-65% predicted). Of the 7 patients with light restrictions, 6 had undergone pneumonectomy before the age of 10 years, and the period between the operation and the time of testing varied between 2 and 12 years. None of the patients was shown to have obstructed airways (forced expiratory volume in 1 second/forced VC% > 70). One patient who underwent a right pneumonectomy displayed an excessive shift of the mediastinum trachea and esophagus 1 year after the operation (Figure 5), but the patient had no findings of compression of the left main bronchus and was free of respiratory symptoms, dysphagia, and reflux.

View larger version (87K): [in this window] [in a new window]   Figure 5. Chest CT scan of an 11-year-old boy shows the excessive shift of the mediastinum to the right hemithorax 1 year after pneumonectomy.

	Discussion

Top Abstract Materials and methods Results Discussion Conclusion References

 
Destroyed lung caused by benign inflammatory lung diseases is an end-stage phenomenon prone to serious complications.² For destroyed lung, pneumonectomy proved to be the most expeditious and effective management for serious complications such as massive hemoptysis, secondary fungal infections, secondary amyloidosis, suppurative infections, and pulmonary-systemic shunting.^2,5

The most common cause of destroyed lung in our patients was bronchiectasis. In 2, the diagnosis was congenital cystic bronchiectasis. The causes of bronchiectasis in children are numerous. Nevertheless, in our patients the most common causative factor was frequent pulmonary infection. Most of our patients had histories of insufficient and irregular antibiotic use. Enlarged parabronchial lymph nodes after pulmonary infections or narrowing secondary to thickening of the bronchial lumen augment the progression of the bronchiectasis with pneumonia and destruction and may lead to total pulmonary bronchiectasis.^1,12,13 The same situation is considered to occur in children with tuberculosis.⁷ There was thickening in the main bronchus in 4 patients as a result of external pressure and intraluminal thickening and narrowing in the main bronchi of 5 patients. Foreign body aspirations can also lead to whole lung bronchiectasis over the long term, as in 1 of our patients.

The destroyed lung is nonfunctional with demonstrable absent perfusion and ventilation.^2,14 Bronchiectasis is commonly complicated by hemoptysis, abscess formation, septicemia, chronic suppurative states, and amyloidosis.¹ The persistence of major inflammatory lung disease, especially pulmonary tuberculosis, presents an evolving global problem.^15,16 The lung is the portal of entry for this disease, and it is the organ most involved pathologically. In the lung, tuberculosis produces an inveterate, necrotizing, granulomatous process that involves the pleura. The destroyed post-tuberculosis lung is a pathologic term describing the complication of end-stage inflammatory residues that follows gross pulmonary destruction by tuberculosis and other causes. Post-tuberculosis acute suppurative secondary infections in destroyed lungs are common. These infections may be bacterial or fungal with significant bronchiectasis or in a residue cavity.^1,5 Surgery in patients with tuberculosis infections is indicated for the treatment of a destroyed post-tuberculosis lung.

Chronic infectious complications in patients with destroyed lung include recurrent low-grade infections and frequent hospitalizations. Chronic and acute hemoptysis can be both debilitating and life-threatening. The severe constitutional symptoms seen in this patient group include fever and inanition.¹ Growth retardation was found in 58.8% of our patients. Improved nutrition was given to all our patients during the preoperative period. Correct anti-tuberculosis treatment should be administered to patients in whom active tuberculosis is diagnosed histologically in the postoperative period. We did not provide anti-tuberculosis chemotherapy to any of our post-tuberculosis patients because we did not identify histologically active tuberculosis in the postoperative period. The presence of active tuberculosis should be examined in the preoperative period and treated before surgery. Two of our patients had pneumonia that rapidly progressed to total lung destruction. The chest x-ray film of 1 of these patients had shown no abnormalities 10 months previously. The other patient reported pulmonary symptoms only 2 months previously. Both patients had a history of severe pulmonary infection and inadequate treatment. Pulmonary infections that are not treated promptly or adequately may lead to the total loss of the lung on 1 side, particularly in children. We cultivated K. pneumoniae from 1 of these patients. There are few studies about pneumonias resulting in necrotizing pneumonia or pulmonary gangrene.^4,17,18 These studies note that in addition to late or inadequate treatment, some patients have histories of aspiration. However, in 3% to 5% of patients, despite adequate therapy, the disease process may progress to irreversible respiratory failure and death.⁴ In such patients, the most common agents are Streptococcus pneumoniae, K. pneumoniae, Bacteroides fragilis, P. aeruginosa, Haemophilus influenza, Staphylococcus aureus, and Escherichia coli.^4,17-19 The enzymes and toxins released by bacteria are the most important factors in the development of rapid lung injury.^4,17,18 A history of aspiration was absent in 2 of our patients.

Left lung involvement was found in 14 of our patients (82.3%), and only 3 patients (17.6%) had right lung involvement. Left lung involvement was also more common in other series.^2,5,13 There are a number of possible reasons for this. The left main bronchus is considerably longer and approximately 15% narrower than the right main bronchus,¹³ and the peribronchial space is limited by its proximity to the aorta; thus, it is more prone to obstruction by the enlargement of adjacent lymph nodes. In addition, the more horizontal course of the left main bronchus, compared with the right main bronchus, may have an effect on the drainage of secretions.^1,13

A bronchus blocker Fogarty embolectomy catheter is used for younger children and in those in whom a double-lumen intubation tube for the drainage of lung secretions cannot be used.²⁰ We used a Fogarty catheter in 2 patients, but it was removed after ventilation difficulties were noted. It was difficult to properly position the catheter. This problem occurred either because of the inexperience of the anesthetist or as a result of the catheter being disturbed when the patient was being moved. Furthermore, Fogarty catheters may become displaced during manipulation of the bronchus, causing spillage or airway obstruction. Some authors advocate using the prone position to avoid contamination of the healthy lung, although a prone thoracotomy is more challenging for both the surgeon and the anesthetist.^7,20,21 We did not use the prone position for surgery because of the difficulty of the position and our inexperience in using it, and because its use may have prolonged the operation. We believe that preoperative aggressive chest physiotherapy, adequate and prolonged preoperative preparation, precise sputum control, frequent intraoperative aspiration, and bronchoscopic aspiration of the side that will be resected before the introduction of the endotracheal tube were sufficient to prevent spillage. We did not encounter intraoperative spillage in any of our patients as a result of our precautionary measures.

Pneumonectomy for inflammatory lung disease is frequently associated with high morbidity rates, and the frequencies of postpneumonectomy space empyema and BPF are high. It is essential to treat underlying infections before surgery in an effort to minimize sputum production, maximize the patient’s nutritional status, minimize the chance of intraoperative spillage, and decrease the risk of postoperative BPFs and postpneumonectomy space empyemas. There is scarce literature available on pneumonectomies in children, with postpneumonectomy complication rates of approximately 20% in the series we located.^22,23 The complication rate in our series was similar at 23.5%. It is reported that postoperative morbidity and BPF are more common in patients with tuberculosis, preoperative empyema, completion pneumonectomy, or right pneumonectomy.^1,3,5 During the postoperative period, BPF developed in only 1 patient. This patient had a history of tuberculosis and had undergone a right pneumonectomy, open-window thoracostomy, and myoplasty. The other patient in whom and empyema developed had undergone left pneumonectomy.

Children and adults have been evaluated together in many reports detailing destroyed lung. In these series, the mortality rate was between 1.2% and 25%.² The mortality rate in our series was 11.7% (2/17). One patient who had undergone pneumonectomy died intraoperatively as the result of respiratory insufficiency, and 1 died on postoperative day 3 as the result of postpneumonectomy pulmonary edema. The remaining lung after pneumonectomy is known to be prone to pulmonary edema that can carry a mortality rate of 100%.²⁴ The mechanism suggested for its development is not totally clear but includes increased capillary pressure, altered endothelial permeability, reduced lymphatic carrying capacity, and overaggressive postoperative fluid therapy.²⁵ Because of the possibility of the latter, postoperative fluid administration should be performed with caution, particularly when considering fresh frozen plasma transfusion.²⁶

Patients underwent follow-up for a median of 5.2 years. One of the patients was lost during the follow-up period, and no major problems were observed in the other patients except for mild pulmonary infections. All of the children were observed to grow and develop normally after pneumonectomy. There was marked herniation of the remaining lung with a mediastinal shift to the opposite side on follow-up chest radiography or CT in all of the patients. Postpneumonectomy syndrome developed in some patients (described mainly in neonates and children when tissue is pliant), especially those who underwent right pneumonectomy. A marked shift and counterclockwise rotation of the mediastinum cause compression of the bronchus between the aorta and pulmonary artery, leading to respiratory distress and even death.^20,24 Numerous methods aimed at prevention and treatment have been described, including the use of expandable silicone or saline-filled prostheses that can be inflated as the child grows.^20,24 We did not use any prostheses for intraoperative prevention in our patients. None of our patients had postpneumonectomy syndrome during the postoperative follow-up period. As in our patients with long-standing inflammatory diseases, a gradual mediastinal shift and lung herniation have developed. As a result of this, the possibility of postpneumonectomy syndrome developing in these patients is weak. In addition, we did not want to use a prosthesis in our patients, because according to Blyth and coworkers,²⁰ a prophylaxis can lead to complications.

In young children subjected to pneumonectomy, the size of the remaining lung increases by partial compensatory overgrowth.^10,27 In the youngest patients (ages 3-14 years), Peters and colleagues²⁸ observed that considerable lung herniation can be associated with low residual air volumes and excellent VC and maximum breathing capacity.

In our subjects, those with a single lung coped well, and according to long-term follow-ups, all of these patients performed routine activities without difficulty. The respiratory function test results, according to the remaining lung’s functional volume, were considered good. A slight degree of restricted respiratory dysfunction was present in a significant number of cases. Similar findings have been reported in other studies.²⁴ Patients undergoing pneumonectomy at a younger age have a better VC than those who undergo the same procedure at an older age. It is believed that the remaining lung is better able to expand in younger patients. This finding is supported by a number of studies that have noted the compensatory growth with hyperplasia in younger children^10,28 and hypertrophy and dilation in older children.¹⁰ Over the long term, the postoperative VC of patients was higher for those who were older and who could perform function tests (Table 2). However, our younger patients had a higher mean VC percent prediction than those who underwent pneumonectomy at an older age. In addition, the exercise tolerance level of the younger children also increased.

Lezama-del Valle and colleagues²⁴ described the long-term complications of scoliosis after pneumonectomy. In children aged 1 to 3 years, scoliosis progressed from 10° to 30° in 3 children in 3 to 5 years. We were unable to locate other studies on the long-term effects of scoliosis in children who had undergone pneumonectomy. We found that 6 of 13 patients demonstrated light (<10°) scoliosis at an average of 5.2 years after pneumonectomy (range 1-12 years). Five of those patients with scoliosis were aged less than 7 years of age at the time of surgery. Patients with light scoliosis do not require treatment, although they should be examined at increasing intervals to establish whether scoliosis has progressed or not.¹¹ In 1 patient with 30° scoliosis, Lezama-del Valle and colleagues²⁴ used a Boston jacket to correct the deformation to 16°. Although patients who underwent pneumonectomy before the age of 10 years have the possibility of developing scoliosis in the long term, we do not think that treatment will be required or that their resting posture will be significantly affected.

	Conclusion

Top Abstract Materials and methods Results Discussion Conclusion References

 
The insufficient and inadequate use of medications for pulmonary infections and tuberculosis in patients, and the lack of follow-up over time, create a background for lung destruction. Destroyed lung describes a totally nonfunctioning lung. When destroyed lung is ascertained, resection should be performed to prevent complications, even though some patients do not report significant symptoms. Patients should be well prepared during the preoperative period in regard to nutritional status and infective process to minimize postoperative complications. Children can easily tolerate pneumonectomy with acceptable rates of morbidity and mortality. In time, the remaining lung expands to compensate for the loss of the removed lung. Children acquire good exercise tolerance and higher lung volumes as the result of partial compensatory lung growth and better nutritional status in the long term. Those who undergo pneumonectomy and have a healthy remaining lung tend to have a regular family, social, and work life. In the short term, patients should not have major skeletal deformation.

	References

Top Abstract Materials and methods Results Discussion Conclusion References

 

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