In Turkey, echinococcosis is an endemic disease, and

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1 Capitonnage Results in Low Postoperative Morbidity in the Surgical Treatment of Pulmonary Echinococcosis Sadik Yaldiz, MD, Soner Gursoy, MD, Ahmet Ucvet, MD, Demet Yaldiz, MD, and Seyda Kaya, MD Division of Thoracic Surgery, Celal Bayar University School of Medicine, Manisa; Dr Suat Seren Chest Diseases and Thoracic Surgery Training Hospital, Izmir; and SB Tepecik Research and Teaching Hospital, Izmir, Turkey Background. The main surgical techniques in the treatment of pulmonary echinococcosis are cystotomy alone, cystotomy and capitonnage, enucleation, and pericystectomy. Controversy persists regarding the selection of surgical technique. We reviewed our experience to identify the impact of capitonnage on outcomes. Methods. A single-institution retrospective analysis was made of the 308 consecutive patients with thoracic hydatid disease treated surgically during 17 years. Results. The most common presenting symptoms were cough and chest pain. At presentation, 69 patients (22.4%) had complicated hydatid disease, cyst rupture into bronchus in 62 and into pleural cavity in 7. Bilateral involvement occurred in 37 patients (12.0%), simultaneous hepatic cysts in 36 (11.6%), and intrathoracic extrapulmonary involvement in 14 (4.5%). Surgery consisted of cystotomy with capitonnage in 271 patients (92.2%), cystotomy and closure of bronchial openings in 20 (6.8%), and lobectomy in 3 (1.0%). Hospital mortality was zero; postoperative complications developed in 21 patients (6.8%). Conclusions. Cystotomy with capitonnage has a low complication rate. Pulmonary resection is best limited to patients with parenchymal destruction secondary to infection. (Ann Thorac Surg 2012;93:962 7) 2012 by The Society of Thoracic Surgeons In Turkey, echinococcosis is an endemic disease, and the annual incidence of hydatid disease is 4.9 cases per 100,000 inhabitants [1]. Surgery remains the treatment of choice for cure of Echinococcus granulosis pulmonary cysts [2]. The goal of treatment in the hydatid cyst is to remove the endocyst and close the pericystic cavity [3]. Capitonnage has long been the practice to avoid postoperative air leak and empyema formation. But in recent years, some investigators suggested that capitonnage does not provide any additional benefit, and that cystotomy only with closure of bronchial openings will be sufficient for the treatment [4 6]. That has led us to retrospectively review our experience of 308 consecutive patients, to further clarify the role of capitonnage in the management of pulmonary hydatid disease. Patients and Methods Accepted for publication Nov 7, Address correspondence to Dr Yaldiz, Kahramandere mah, 758 sok, Onurcan sitesi, No. 23 Guzelbahce, Izmir, Turkey; msyaldiz@gmail.com. The cases of 308 consecutive patients with pulmonary hydatid disease in a single institution between January 1993 and December 2009 were retrospectively reviewed. The study was carried out in agreement with Turkish laws on biomedical research and according to the principles outlined in the Helsinki agreement. All patients received full information and signed an informed consent form. The study group consisted of 163 male patients (53%) and 145 female patients (47%) whose mean age was years (range, 8 to 76 years). The preoperative evaluation of all patients was carried out by means of a physical examination, chest roentgenogram, abdominal ultrasonography, and computed tomography of the chest and upper abdomen. Fiberoptic bronchoscopy was only carried out if a mass lesion, rather than a hydatid cyst, was suspected preoperatively. When the pulmonary hydatid cyst had ruptured into the bronchus or pleural cavity, with or without infection, it was defined as a complicated cyst. The decision as to which cyst should be removed first was based on the susceptibility of each to rupture, their size, and the risk of dissemination. For patients in whom both an intact and a ruptured cyst were seen, the intact cyst was treated first. Most patients had a double-lumen endotracheal tube. The surgical approach was posterolateral thoracotomy (standard or muscle sparing), and for bilateral hydatidosis, median sternotomy, or a 2-month interval staged thoracotomy. Patients with right pulmonary and liver dome cysts were treated by right thoracophrenotomy. All other hepatic cysts were treated in the general surgery department. Because of recurrence, 7 patients underwent rethoracotomy. The preferred surgical treatment procedure was cystotomy with capitonnage. Cystotomy and closure of the bronchial openings without capitonnage were performed only for the cysts located in the lower lobes and facing down to the diaphragm. Additional 2012 by The Society of Thoracic Surgeons /$36.00 Published by Elsevier Inc doi: /j.athoracsur

2 Ann Thorac Surg YALDIZ ET AL 2012;93:962 7 CAPITONNAGE LEADS TO LOW POSTOPERATIVE MORBIDITY 963 Fig 1. The germinative membrane after cystotomy. surgical procedures such as small wedge resections, decortication, and debridement of necrotic tissues were also used. Enucleation and pericystectomy were not performed in this study. Lobectomy was carried out only when the cyst was associated with pulmonary parenchymal destruction. Unlike excision, capitonnage refers to emptying the cyst and then closing it by applying sutures so that they approximate the opposing surfaces of the cavity. In the surgical technique, the wound and lung, apart from the area containing the cyst, were covered with sponges moistened with 3% saline solution to prevent the implantation of the scolices or daughter cysts. Owing to risks of leakage into the bronchial system and postoperative tracheobronchial irritation or pulmonary edema, no scolicidal agent was injected into the cyst. The cyst contents were evacuated by needle aspiration. The most prominent part of the cyst was opened (cystotomy; Fig 1). The germinative membrane was removed with ring forceps. The pericystic cavity (Fig 2) was irrigated with saline solution and cleaned with sponges containing diluted povidone-iodine (10%). The leaks were diagnosed by positive ventilation, secured with 2 0 nylon Fig 3. The pericystic cavity obliterated with capitonnage. stitches, and their closure was again reconfirmed by positive ventilation. After the bronchial openings were closed, the residual cavity was obliterated with separate pursestring sutures that were placed carefully to the foldings of pericystic tissue from the deepest level to the surface (capitonnage; Fig 3). The lung was inflated to check for the air leakage (Fig 4). In each patient, two chest tubes were positioned, posteriorly and anteriorly. Postoperatively, air leaks persisting for more than 7 days were defined as prolonged air leaks. When the prolonged air leaks were not recovered within 2 or 3 weeks, it was considered bronchopleural fistula. Hemorrhage causing reexploration, bronchopleural fistula, and empyema were considered major complications. Minor complications were atelectasis, prolonged air leakage, and pneumonia. Operative mortality was defined as death from any cause during hospitalization time within 30 days of the operation. Preoperative anthelmintic treat- Fig 2. The pericystic cavity. Fig 4. The fully inflated lung at the end of the operation.

3 964 YALDIZ ET AL Ann Thorac Surg CAPITONNAGE LEADS TO LOW POSTOPERATIVE MORBIDITY 2012;93:962 7 Table 1. Symptoms Symptom Rate (%) Cough 58 Chest pain 42 Expectoration of salty sputum and fragments of 13 laminated membrane Anaphylactic reaction symptoms 0.3 Blood-streaked sputum 11 Mucopurulent sputum 13 Asymptomatic 22 ment was not used in any patients. Postoperatively, 119 patients (38.6%) who had complicated or multiple hydatid cysts, or both, were placed on albendazole anthelmintic therapy (800 mg daily for adults, 10 mg/kg daily for children) for at least 3 to 6 months. All recurrent hydatid patients underwent rethoracotomy. Results Assessment of clinical findings, chest roentgenograms, thoracic computed tomography, and thoracic ultrasonography led to the correct preoperative diagnosis of pulmonary hydatid disease in 270 patients (87.7%). The other 38 patients (12.3%) were diagnosed intraoperatively. In patients with intact lung cysts, 29% were asymptomatic with incidental diagnosis on routine chest radiograph. The remaining patients exhibited one or more symptoms, the most frequent symptoms being cough and chest pain (Table 1). Fourteen patients (4.5%) presented with extrapulmonary but intrathoracic location of the disease (diaphragm, chest wall, pleura, pericardium, and mediastinum). One hundred twenty-one patients (41.2%) had a solitary lung cyst in the right lung, and 98 patients (33.3%) had one in the left lung only, whereas the remaining 75 (25.5%) were found to have multiple cysts in one or more lobes of one or both lungs. Unilateral multifocal cysts were detected in 38 patients (12.9%) and bilateral involvement in 37 patients (12.6%). Cysts were Table 2. Operative Methods Procedure (n 308) Rate (%) Lung (n 294) Cystotomy with capitonnage Cystotomy without capitonnage Lobectomy Additional surgical procedures a Pericystectomy Enucleation Intrathoracic extrapulmonary (n 14) Total cyst excision Liver (n 12) b Cystotomy with capitonnage a Small wedge resection, decortication, and debridement of necrotic tissues. b Liver dome cysts only. Table 3. Postoperative Complications Complication (n 21) Rate (6.8%) Prolonged air leak Prolonged air leak with empyema Atelectasis Pneumonia Superficial wound infection Hemorrhage causing reexploration Bronchopleural fistula Lung collapse Repeat thoracotomy Pneumothorax Sepsis suppurative in 42 patients (13.6%); 36 patients (11.6%) had cysts larger than 10 cm in diameter. Simultaneous hepatic and pulmonary involvement was seen in 36 patients (11.6%). Twelve patients (3.9%) having concomitant cysts in the liver dome were managed by right phrenotomy. In 4 of these cases, cysts in lower right lung lobe were connected to hepatic cysts with passing through the diaphragm, resembling an hourglass. Sixty-nine patients (22.4%) had at least one complicated cyst. Seven patients (2.3%) had a cyst or cysts opened to the pleural space and presented with pneumothorax and empyema thoracis. These patients all required closed-chest tube drainage initially, and further surgical procedures including lung decortication completed the surgical procedure after their general condition had stabilized. Cyst rupture into a bronchus occurred in 62 patients (20.1%). Expectoration of salty sputum consisting of mucous hydatid fluid and some fragments of the laminated membrane were observed in 41 cases (13.3%). In 271 (92.2%) of 294 pulmonary hydatid patients, the specific surgical treatment for pulmonary hydatid cysts was cystotomy with capitonnage (Table 2). Cystotomy alone with closure of bronchial openings was performed in 20 patients (6.8%). Only 3 patients (1.0%) underwent lobectomy. Additional surgical procedures such as wedge resection, decortication, and debridement of necrotic tissues were carried out in 27 patients (8.7%). Enucleation and pericystectomy was not carried out in any patient. There was no postoperative mortality. Twentyone patients (6.8%) had complications during the postoperative period (Table 3). Complications were as follows: prolonged air leak in 10 patients (3.3%), prolonged air leak with empyema in 2 patients (0.6%), atelectasis in 4 (1.3%), pneumonia in 3 (1.0%), and superficial wound infection in 2 (0.6%). Patients with a prolonged air leak were managed by continuous negative aspiration. Atelectasis was resolved through bronchoscopic aspiration. Patients with pneumonia and empyema were treated with the appropriate antibiotic agents and thoracic drainage. Wound infection was successfully managed with local treatment. No patient underwent a rethoracotomy for the complications.

4 Ann Thorac Surg YALDIZ ET AL 2012;93:962 7 CAPITONNAGE LEADS TO LOW POSTOPERATIVE MORBIDITY 965 Mean follow-up time was 6.3 years (range, 1 to 16 years). In all, 44 patients were lost to follow-up. Recurrence was observed in 7 (2.6%) of 264 patients who were followed up (85.7%). Two patients who did not receive albendazole treatment, and who had intact cysts at the time of initial surgery, had a recurrent cyst develop within the same lobe. Three patients who had a ruptured cyst preoperatively and received albendazole treatment postoperatively had a recurrent cyst develop in a different lobe on the same side. These 5 patients had a second thoracotomy, and cystotomy with capitonnage was performed again. One patient had a pleural recurrence. Recurrence was probably the result of inadvertent spillage of cystic contents into the pleural space during needle aspiration of the cyst at the first operation. This patient was also administered albendazole treatment postoperatively. Second thoracotomy and cystectomy were performed. One patient who did not receive albendazole treatment showed extrathoracic organ recurrence, being the hepatic involvement. It was successfully excised at laparotomy. We have administered albendazole treatment to 119 complicated and multiple cases (38.6%) in the postoperative period for at least 3 to 6 months. The follow-up was completed for 105 of them, with recurrences in only 4 patients. Comment The period of initial growth of primary hydatidosis is frequently asymptomatic. Symptoms may only occur when they grow to a certain size or when complications arise [7]. It has been reported that 8% to 32% of patients are asymptomatic at the time of diagnosis [8, 9]. In our study, the rate of asymptomatic patients was 22.4% in all patients, and 29% in those with intact lung cysts. Most common presenting symptoms were cough and chest pain. A rupture into the pleural cavity usually causes pneumothorax, pleural effusion, or empyema, and is reported as 0.5% to 18.2% in the literature [10]. In 7 (2.3%) of our patients, pneumothorax and empyema developed after rupture into the pleural cavity. They were all managed first by tube thoracostomy, and operated on later. The incidence of anaphylaxis (generalized rash, high fever, pulmonary congestion, and severe bronchospasm) is very low (0% to 0.3%) [11] and was seen only in 1 of our patients (0.3%). Chest roentgenography, accompanied by computed tomography, is the most often used radiologic procedure in diagnosis. However, it is not always possible to diagnose hydatid cyst by chest roentgenogram and computed tomography because of the solid density of an infected hydatid cyst; thus, 12.3% of our patients were diagnosed intraoperatively. The goal of treatment of the hydatid cyst is to eradicate the parasite, remove the endocyst without allowing intraoperative seeding, and to close the pericystic cavity, with maximum preservation of lung tissue [10 13]. Most authors agree that all attempt should be made to remove as little lung tissue as possible, and that resection of pulmonary parenchyma is only indicated when the adjacent tissue is seriously damaged or infected, or when the atelectatic areas are presumably irrecoverable [9]. In the great majority of the current patients (92.2%), a cystotomy with capitonnage was used. The concept of capitonnage is to avoid bronchopleural fistula or abscess formation in the residual cavity [14]. It has been claimed that closure of the cavity with capitonnage can cause atelectasis by obliterating the major bronchi surrounding the cyst cavity and present a potential risk of pulmonary parenchymal distortion. However, Kosar and colleagues [15] have reported that they saw only one atelectasis in 37 patients with hydatid cysts who had undergone capitonnage. Erdogan and colleagues [6] also have reported that they saw no atelectasis in their series of 44 patients. In the current paper, capitonnage was employed in 271 patients (92.2%), and atelectasis was revealed only in 4 patients (1.3%). We think that if capitonnage technique is applied carefully to the foldings of pericystic tissue, then it will cause a very low rate of atelectasis and distortion. The noncapitonnage method is reported as an alternative by some authors [4 6]. In our department, the noncapitonnage method was only performed for the lesions facing to the diaphragm. Owing to the great risk of perforation and spillage of cystic contents into the operative field, enucleation was not carried out in this study. Although pericystectomy is sometimes recommended for treatment, we did not prefer this technique because the pericyst is not parasitic, so excision in its entirety is not necessary and may cause complications such as severe air leak. The surrounding lung tissue is atelectatic but often not infected, and after cyst removal, it usually reexpands well. A ruptured cyst was considered to be infected when the patient exhibited accompanying problems of purulent sputum, leukocytosis, fever, and pericystic pneumonitis with or without lobar and segmental pneumonia. Even in these cases, lobectomy was not performed in this study. It is reported that regardless of size, giant hydatid cysts also could usually be surgically treated without lung resection, and cystotomy with capitonnage was the most commonly performed operative method [16]. The rate of lobectomy is stated as 0.5% to 45% in the published reports [13]. In a period of 17 years, only 3 of our patients (1.0%) with completely destroyed lobe underwent lobectomy. In most series, mortality is reported to be 0% to 2.3% [17]. There was no in-hospital mortality in our series. The postoperative complication rate of hydatid disease ranges between 1% and 39% [3, 8, 18 20], and prolonged air leak was the most frequently encountered complication. We have observed prolonged air leak in 10 patients (3.3%), 7 of whom were the patients without capitonnage. It is reported that the time of air leak was longer postoperatively in the group without capitonnage than in the group with capitonnage, even if bronchial leaks were not detected during surgery [21]. Opposing this, it is suggested that after the closure of the bronchial openings, capitonnage is not necessary for the surgical treatment of pulmonary hydatid cysts, but one of their patients with a bronchopleural fistula was treated with the thoracoplasty technique 6 months after the primary surgical therapy, in

5 966 YALDIZ ET AL Ann Thorac Surg CAPITONNAGE LEADS TO LOW POSTOPERATIVE MORBIDITY 2012;93:962 7 the noncapitonnage group [6]. Major complications are reported in a study without capitonnage, foremost being the lung collapse (11%), empyema (14%), repeat thoracotomy (7%), and sepsis (1.4%) [19]. Despite all attempts, it may be rather difficult to identify and close all of the bronchial openings because of visual obstruction of blood clots and secretion. Thus, capitonnage provides the complete closure of the cavity, and acts as a second barrier against prolonged air leak in the prevention of bronchial air leak [15]. Postoperative air leak after capitonnage must be considered as a technical error. In the early postoperative period, chest radiographs of the patients undergoing capitonnage generally showed a consolidated area at the operation field; but radiological control after 2 months showed normal radiological findings or a thin fibrotic band, suggesting the cavity was fully obliterated. Preoperative medical treatment was not used in the current series because anthelmintics weaken the cyst wall, thus increasing the likelihood of cyst rupture. Wen and Yang [22] found a 77.3% incidence of cyst rupture in 21 patients with hydatid disease who were treated with albendazole. Dogan and coworkers [8] administered mebendazole treatment to 28 patients in the preoperative period. During this treatment, 4 cases were urgently operated on owing to massive hemoptysis and 6 cases were reported to have cyst rupture into the bronchus. Only 3 cases showed regression of the cyst [8]. Generally, drug treatment is recommended postoperatively for complicated, multiple, and recurrent pulmonary hydatid cysts. In this study, the follow-up was completed in 105 patients who were put on a regimen of albendazole treatment, and recurrence was seen in only 4 of them. Lung-conserving procedures are optimal with pulmonary hydatidosis, but video-assisted thoracic surgery is strongly recommended for selected patients [23]. It is possible to remove the cystic membrane thoracoscopically, but uncontrolled spillage of cyst contents may cause anaphylactic reaction or later development of the new cysts in the contaminated tissues [24]. Therefore, we did not perform this procedure in our unit. Although there is not yet consensus regarding the surgical procedure that might be used, we believe that open surgery involving removal of the cyst membrane, closure of the bronchial openings, and capitonnage results in limited postoperative complications and a low recurrence rate and low mortality. A randomized trial to critically evaluate the value of capitonnage in the management of residual cavity remains a priority. References 1. Kilic D, Kaya I, Kamas A. Health statistics. Ankara, Turkey: Republic of Turkey Ministry of Health, Research Planning Coordination Council, 2002; World Health Organization (WHO) Informal Working Group on Echinococcosis. Guidelines for treatment of cystic and alveolar echinococcosis. Bull WHO 1996;74: Kavukcu S, Kilic D, Tokat AO, et al. Parenchyma-preserving surgery in the management of pulmonary hydatid cysts. J Invest Surg 2006;19: Eren MN, Balci AE, Eren S. Non-capitonnage method for surgical treatment of lung hydatid cysts. 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