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Ann Thorac Surg 2002;74:889-892
© 2002 The Society of Thoracic Surgeons


Original article: general thoracic

Ruptured hydatid cysts of the lung in children: clinical review and results of surgery

Akin Eraslan Balci, MD*a, Nesimi Eren, MDa, Sevval Eren, MDa, Refik Ülkü, MDa

a Department of Thoracic and Cardiovascular Surgery, Dicle University School of Medicine, 21280 Diyarbakir, Turkey

Accepted for publication May 13, 2002.

* Address reprint requests to Dr Balci, Department of Thoracic and Cardiovascular Surgery, Dicle University School of Medicine, 21280, Diyarbakir, Turkey
e-mail: abalci{at}dicle.edu.tr


    Abstract
 Top
 Abstract
 Introduction
 Material and methods
 Results
 Comment
 References
 
Background. Rupture of a hydatid cyst may cause some unique problems, especially in children.

Methods. Sixty-three children with a total of 68 ruptured lung hydatid cysts were operated on between 1980 and 2000. Mean age was 12.3 years (range, 1 to 15 years). Radiographic findings were hydropneumothorax (20.6%) and air-fluid level (19%). Mean follow-up was 19.3 months.

Results. Transthoracic needle aspiration was responsible for the rupture in 3 children. The interval between cyst rupture and operation was less than 24 hours in 10 patients (15.9%), 1 to 4 days in 36 (57.1%), and more than 4 days in 17 (27%). Resection rate was 22.1%. The most frequent operative method was cystotomy and capitonnage (38%). Morbidity was 25.4% (extended air leak 5, empyema 3, bronchopleural fistula 3, atelectasis 3, pneumonia 2). Mortality was 4.7% (hemoptysis 1, pneumonia and sepsis 1, aspiration of hydatid material 1). Morbidity and mortality seem to be more frequent in late cases.

Conclusions. Early surgical intervention with single-lung ventilation and maximum parenchyma preservation are recommended.


    Introduction
 Top
 Abstract
 Introduction
 Material and methods
 Results
 Comment
 References
 
Echinococcal cysts always carry the risk of rupture with anaphylactic potential, and the risk of spillage to healthy viscera. Many factors may be responsible for the rupture, such as trauma or coughing increasing the intrathoracic pressure or expansion of the cyst size. No correlation had been found between cyst size and intracystic pressure [1].

There are several reports about hydatid cysts in children [25] but none especially about ruptures, if one excludes those about complications such as rupture into the pleura [6], tension pneumothorax [7], or airway obstruction [8]. The purpose of this study was to document the specifications of ruptured cysts in children.


    Material and methods
 Top
 Abstract
 Introduction
 Material and methods
 Results
 Comment
 References
 
Between 1978 and 2000, 90 children with ruptured hydatid cysts of the lung and 236 children with intact cysts were operated on. There were 63 children with pure ruptured cysts; we excluded the children with intact cysts associated with ruptured ones (14 cases) and those with other organ cysts (13 cases). These 63 children constitute the study group. The symptoms, localizations, and radiologic findings of the cysts and the indications, specifications, and results of the operations were studied. The mean age of the study group was 12.3 years (range, 1 to 15 years), and the age group distribution was as follows: 0 to 4 years, 21%; 5 to 9 years, 30.2%; and 10 to 14 years, 48.8%. The male to female ratio was 2.3. Initial data were collected retrospectively from the patients’ files. After the operation, within 6 months, all of the patients had been examined radiologically at least once in a policlinic. For the study, data were obtained from patients themselves or from relatives by mail or calling. Mean follow-up was 19.3 ± 11 months (range, 9 to 31 months) for all patients. The proportion of patients followed up for more than 1.5 years was 47%.

Most frequent respiratory symptoms were cough in 23 (36.5%) and fetid expectoration in 23 (36.5%; Table 1). All patients had a chest radiograph. An image on the chest roentgenogram with a very round appearance and decreasing density from the center to periphery was known as pathognomonic for intact lung hydatid cyst, especially for the endemic region. In conditions including turbid intracystic fluid, degenerated germinative membrane, or calcification or gross infection of the cyst, it was considered to be ruptured. Radiograph findings of the 63 children were hydropneumothorax in 13 (20.6%), air-fluid level mimicking lung abscess in 11 (17.5%), total pneumothorax in 11 (17.5%), air cysts in 12 (19%), localized empyema in 9 (14.3%), and water lily sign in 7 (11.1%). Other diagnostic methods used were thoracic and liver ultrasonography in 34 patients (54%), computed tomography in 38 (60.3%), and thoracentesis in 12 (19%). Because of their lack of specificity, Weinberg and Casoni tests were not used. Blood tests showed eosinophilia in most patients. On the other hand, no pathognomonic radiologic or serologic findings were found to establish a clear perforated hydatid cyst diagnosis. Any acute symptom (such as cough, hemoptysis, fever, chest pain, vomiting, or membrane expectoration) or sudden aggravation in a present symptom was referred to as cyst rupture.


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Table 1. Symptoms of Perforated Lung Hydatid Cysts of Childrena

 
The main operative indication was deterioration or no improvement in the general condition in spite of antibiotic therapy, chest physiotherapy, and fetid or bloody expectoration in a patient with a lung lesion suggesting a perforated cyst. A lack of improvement in the radiologic lesion with time and a patient coming from an endemic region were other supporting factors. The reasons for the thoracotomy were fever in 20 patients (31.7%), hemoptysis in 12 (19%), dyspnea in 9 (14.3%), history of membranous expectoration in 8 (12.7%), computed tomographic findings in 5 (7.9%), exploration in 3 (4.7%), clear water aspiration from former thoracentesis in 3 (4.7%), and membranous hydatid material observed in the chest tube of patients misdiagnosed as having empyema in 3 (4.7%).

The primary method for repairing the part of the lung with the perforated cyst was to close the bronchial openings after removing the germinative membrane, if present, and capitonnage. Median sternotomy was not used because of the risk of infection. For cysts that ruptured to the bronchus, rather than to the pleura, a cystotomy was required to visualize the cystic cavity. The lung tissue next to the cyst was observed and evaluated with ventilation of the lung. If the parenchyma was hepatized and not ventilated with insufflations by the anesthesiologist, a resection was considered. Indications for resection were advanced parenchyma destruction resulting from infection or compression of the cyst to the lung. Determining the extent of the resection was related to intraoperative findings and the surgeon’s opinion. Thus, if the lesion involved at least two thirds of the lobe, a lobectomy was performed. No stapler was used. In empyema cases (5 children) with trapped lung caused by pleural peel, a decortication was performed to reexpand the lung.

All patients received antibiotic therapy in the preoperative and postoperative periods. Mebendazole or albendazole was used postoperatively as an antihelmintic, but not routinely, because of the high dose needed, the lack of a proven effect on hydatid disease and poor patient tolerability. There was no standard protocol for antibiotics such as anthelmintics. In general, first-generation and second-generation cephalosporins were empirically used if the special culture was not available. Any air leak continuing more than 10 days was considered prolonged or extended. Atelectasis was diagnosed with postoperative roentgenograms and decreased respiratory sounds by auscultation associated with a sudden increase in body temperature.


    Results
 Top
 Abstract
 Introduction
 Material and methods
 Results
 Comment
 References
 
There were 68 ruptured cysts in the 63 patients: in the right lung in 36 (53%), in the left lung in 27 (39.7%), and in the pleura in 5 (7.3%). Table 2 shows definite localizations. Three patients had two cysts, 1 patient had three, and the remaining patients had one. There were 2 children with bilateral ruptured cysts; they underwent staged thoracotomy with a 7-day interval and used a double-lumen endotracheal tube.


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Table 2. Location of Perforated Cystsa

 
The lung resection rate was 22.1%, with lobectomy being the main method used (12.7%). Indications for resection were advanced parenchymal destruction resulting from infection or compression of the cyst to the lung. No stapler was used. The most common operative method was cystotomy and capitonnage (38%), followed by cystotomy alone (25.4%). Table 3 summarizes the operations. In 5 children with localized empyema, decortication was performed to remove the visceral peel that trapped the lung. Parietal peel was removed completely in 2 and incompletely in 2 and not removed in 1. In 4 other patients, no additional intervention was performed other than freeing the lung during the surgical procedure.


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Table 3. Operative Methods for Perforated Hydatid Cystsa

 
Morbidity occurred in 16 children (25.4%). The most frequent complication was extended air leak (7.9%). Table 4 shows the complications. All 3 patients with empyemas were treated with chest tube initially. One of them underwent decortication 6 weeks after the cyst operation. Bronchopleural fistula formation occurred in 2 patients who underwent cystotomy and capitonnage. Conservative treatment with drainage, respiratory exercise, and antibiotic therapy was sufficient; no rethoracotomy was needed. Bronchoscopic aspirations and respiratory exercises were successful in reexpanding the lung in all cases of atelectasis. All children who had postoperative complications were operated on more than 4 days after the rupture, except 2. One of them was operated on 1 day after and the other 2 days after, and both had extended air leaks. The morbidity rate of patients operated on 4 days after the rupture was 82.3% (14 of 17), and 4.3% (2 of 46) in patients operated on earlier. Morbidity was more frequent in late cases (p < 0.05, with {chi}2 test), probably resulting from infection.


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Table 4. Serious Postoperative Complications of Perforated Hydatid Cysts

 
Mortality was 4.7%: 3 children died. A 7-year-old girl died on the fourth postoperative day as a result of hemoptysis. An 11-year-old boy died on the 17th day as a result of pneumonia and sepsis. The third child had undergone intraoperative bronchial aspiration for acute obstruction of the airways resulting from aspiration of ruptured hydatid cyst material. This 9-year-boy died on the 11th day of mechanical ventilation. After the last death (1993), single-lung ventilation was used routinely. Excluding the last case, operated on in the first 24 hours, all had been operated on more than 4 days after the rupture. Therefore, only 1 patient was lost from those operated on early. The mortality rate of the patients operated on late was 11.7% (2 of 17), and 2.1% in early operations. There were no subsequent late complications or recurrence.

In patients who received separate lung ventilation (19 cases), there was no mortality and there were only complications that needed no or relatively less invasive intervention. There was no correlation between operation type and complications or mortality.

Recurrent transthoracic needle aspirations performed during diagnosis in another hospital were responsible for the rupture in 3 children. No specific cause was identified in the remaining children.

The interval between cyst rupture and operation was less than 24 hours in 10 patients (15.9%), 1 to 4 days in 36 (57.1%), and more than 4 days in 17 (27%).

The period of postoperative antibiotic treatment (mean, 3.6 days; range, 2 to 10 days) was adjusted according to the clinical findings of the patients.


    Comment
 Top
 Abstract
 Introduction
 Material and methods
 Results
 Comment
 References
 
In children, the presence of hydatid disease in the lung has been reported to be up to 67% [9, 10], whereas in adults cysts are more prominent in the liver. The ratio of ruptured cysts to intact ones in children was 26.7% in our series; however, it may be up to 47% [3, 11]. Whereas intact cysts may exhibit minimal symptoms or be asymptomatic, perforated cysts are always symptomatic. Determining the rupture time, however, was not difficult in our patients.

We observed more complications and higher mortality in cysts operated on late. In addition, some symptoms, especially fever, were resistant to medical treatment with antibiotic and analgesic or antipyretic drugs. Surgery was the only method that effectively reduced the fever. The same antibiotics were used postoperatively and stopped generally within 2 to 4 days. It may be speculated that infection with fever may lead to pneumonia and sepsis. In other series, as in ours, complication and resection rates, as well as mortality, have been higher in complicated cysts [11, 12]. In the pediatric age group, because of the low resistance of the lung to cysts, expansion may cause a tension pneumothorax as a result of the rapid growth of the cyst, advocating early surgical removal of any suspected cyst to avoid further complication [7]. The mortality rate was as high as 4.2% in some series that included both complicated and uncomplicated cysts [13]. We have to emphasize that, like intact cysts, ruptured ones must be operated on as soon as the diagnosis is established, in elective conditions (not emergency), and that separate lung ventilation is important for decreasing morbidity and mortality. The better cases in our series involved children operated on before 4 days and with separate lung ventilation.

Complicated hydatid cysts, especially those that ruptured into the pleura, are difficult to diagnose radiologically. In 3 patients, cysts had ruptured into the pleura but these were misdiagnosed as empyema (roentgenogram, computed tomography, and ultrasonography had been performed). After chest tube insertion, hydatid material was observed within the tube, and thoracotomy was performed. Although computed tomography has been reported to be currently the most sensitive diagnostic tool for demonstrating liver hydatid cyst rupture [14], further studies may be useful to elucidate specific radiologic signs for diagnosing ruptured lung hydatid cysts. In complicated (ruptured, infected) or undiagnosed cases, all the findings are suggestive, not diagnostic. Although the computed tomographic diagnosis of ruptured pulmonary hydatid cyst was the most difficult [15] because of infection, in the planning of the operation it was successful in detecting the localization, contents, and borders of the lesion.

Medical treatment of intact cysts is not effective, but may be useful for ruptured ones. Our patients did not take any antihelmintic drug before the operation. There was no recurrence during the follow-up period. Transthoracic needle aspiration may cause rupture of cysts and spillage of their contents; hydatid fluid with scolices and daughter cysts could cause severe anaphylactic reaction or later development of new cysts in the contaminated tissues. In 3 children, needle aspiration was responsible for the rupture. We advise against puncture when a hydatid cyst is suspected because this may result in rupture, which in turn leads to more complications. In intact cysts, our resection rate was 7% versus a relatively high rate of ruptured ones (22.1%). This high resection rate for ruptured cysts is wholly because of the complication of hydatid disease (rupture), which causes significant destruction of the lung tissue. Similarly, morbidity and mortality are also less in intact cysts (14.4% and 1.5%, respectively). Only the recurrence rate is higher in intact ones (2.5% versus 0%), probably not only because of successful operation but also because of ruptured or infected cysts having a much lower reinfection capacity.

Bronchi opening into the pericyst cavity allow the discharge of fluid matter but not the escape of solid remnants of the collapsed parasite in patients with ruptured lung hydatid cysts [8]. Operative manipulation of chronic ruptured pulmonary hydatids can force fragments of the laminated membrane or small daughter cysts into the bronchial tree, resulting in acute obstruction of airways and the need for intraoperative bronchial aspiration [8]. We have encountered these complications in some adults and 9 children, of whom 5 had intact cysts and 4 had ruptured cysts. All exhibited respiratory failure with a decrease in arterial oxygen pressure or saturation, ventilation difficulty during operation, and prolonged ventilatory support in the postoperative period. Two children, one with an intact cyst and the other with a ruptured cyst, died on the third and fifth postoperative days, respectively, of respiratory failure. We used continuous positive airway pressure delivered by an anesthesiologist during cystotomy routinely to prevent the escape of cyst content to the bronchi. This method may be useful but is not sufficient and dependable because the duration of positive pressure is time-limited and excessive pressure produced by surgical manipulations rather than by ventilation can cause escape of hydatid fluid into the bronchial tree. In addition, before the cystotomy or other surgical method, cyst rupture can occur and force fragments into the tree. Thus, we prefer a double-lumen intubation tube or ordinary cuffed tracheal tube introduced into the main bronchus of the healthy side and single-lung ventilation. No similar complications have been caused by single-lung ventilation in recent years, possibly because of better anesthesiologic and postoperative facilities.

Surgical treatment is effective in children with complicated cysts as well as uncomplicated ones, with low mortality and no late complications or recurrence for this relatively high-risk group. Operation must be performed early because of the high complication rate in patients who were operated on late. The choice of surgical technique depends on the conditions encountered during the operation. When parenchymal destruction is present, resection must be performed. Only the destroyed parenchyma should be removed. Under other conditions, cystotomy and capitonnage are the main surgical methods. In cases of trapped lung caused by infection (empyema), decortication of the pleural peel must be performed. The type of operation was not correlated with morbidity or mortality. Single-lung ventilation is preferred to prevent aspiration of cystic material and respiratory complications, and, in turn, morbidity and mortality. Although postoperative morbidity seems to be high, perforation or infection were responsible rather than the operation itself.


    References
 Top
 Abstract
 Introduction
 Material and methods
 Results
 Comment
 References
 

  1. Yuksel M., Kir A., Ercan S., et al. Correlation between sizes and intracystic pressures of hydatid cysts. Eur J Cardiothoracic Surg 1997;12:903-906.[Abstract]
  2. Solak H., Yeniterzi M., Yüksek T., et al. The hydatid cyst of the lung in children and results of surgical treatment. Thorac Cardiovasc Surg 1990;38:45-47.[Medline]
  3. Topçu S., Kurul C., Tautepe ., et al. Surgical treatment of pulmonary hydatid cysts in children. J Thorac Cardiovasc Surg 2000;120:1097-1101.[Abstract/Free Full Text]
  4. Celik M., Senol C., Keles M., et al. Surgical treatment of pulmonary hydatid disease in children: report of 122 cases. J Pediatr Surg 2000;35:1710-1713.[Medline]
  5. Mutaf O., Arikan A., Yazici M., et al. Pulmonary hydatidosis in children. Eur J Pedaitr Surg 1994;4:70-73.
  6. Cernay J., Bensenouci A., Boukhelal H., et al. The rupture of the pleura: a serious complication of hydatid cysts of the lung in children. Rev Fr Mal Respir 1979;7:45-47.[Medline]
  7. Stewart M.P., Cunningham M.S. Tension pneumothorax complicating pulmonary hydatid disease in childhood. Clin Pediatr (Phila) 1987;26:422-424.
  8. Saidi F., Rezvan-Nobahar M. Intraoperative bronchial aspiration of ruptured pulmonary hydatid cysts. Ann Thorac Surg 1990;50:631-636.[Abstract]
  9. Tsakayiannis E., Pappis C., Moussatos G. Late results of conservative surgical procedures in hydatid disease of the lung in children. Surgery 1970;68:379-382.[Medline]
  10. Slim M.S., Akel S.R. Hydatidosis in children. Prog Pediatr Surg 1982;15:119-129.[Medline]
  11. Doan R., Yüksel M., Çetin G., et al. Surgical treatment of hydatid cysts of the lung: report on 1055 patients. Thorax 1989;44:192-199.[Abstract/Free Full Text]
  12. Dhaliwal R.S., Kalkat M.S. One-stage surgical procedure for bilateral lung and liver hydatid cysts. Ann Thorac Surg 1997;64:338-341.[Abstract/Free Full Text]
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