Echinococcus granulosus is frequently encountered in

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1 Surgical Treatment of Pulmonary Hydatid Cysts: Is Capitonnage Necessary? Akif Turna, MD, Muhammet Ali Yılmaz, MD, Gökhan Hacıibrahimoğlu, MD, Cemal Asım Kutlu, MD, and Mehmet Ali Bedirhan, MD Department of Thoracic Surgery, Yedikule Hospital for Chest Disease and Thoracic Surgery, Zeytinburnu, Istanbul, Turkey Background. Hydatid disease of the lung caused by Echinococcus granulosus is frequently encountered in Mediterranean countries. The ideal surgical method for treating this disease is still unknown. Methods. Between 1994 and 2001, 71 patients with pulmonary hydatid cysts were treated surgically. There were 41 male and 30 female patients with a mean age of 30.2 years (range, 5 to 70 years). Cystotomy and closure of bronchial openings were performed in all patients. Obliteration of the residual cavity by imbricating sutures from within (capitonnage) was achieved in 39 patients (group 1). There were 34 patients with intact cysts and 37 patients with at least one complicated cyst. The average diameter of the cysts was 6.4 cm, and the mean number of cysts per patient was 1.4. The surgical outcome was assessed in group 1 patients and in patients who had undergone closure of bronchial openings without capitonnage (group 2; n 32). The groups were comparable in regard to clinical characteristics. Results. There was no mortality. The total hospitalization time (mean standard error of the mean) was days for group 1 and days for group 2 (p 0.91). Stay in the intensive care unit was days in group 1 and days in group 2 (p 0.90). The duration of air leak was days in group 1 and days in group 2 (p 0.87). There was no significant difference between groups in the development of empyema (1 patient in group 2 only) and prolonged air leak (5 patients in group 1 and 4 in group 2). There was also no significant difference in the rate of recurrence (3 patients in group 1 only). Conclusions. We conclude that capitonnage provides no advantage in operations for pulmonary hydatid cysts. (Ann Thorac Surg 2002;74:191 5) 2002 by The Society of Thoracic Surgeons Echinococcus granulosus is frequently encountered in sheep- and cattle-raising regions of the world and has been observed most often in Australia, New Zealand, South Africa, South America, and Mediterranean countries [1 3]. Humans act as accidental intermediate hosts and harbor cysts, which are most commonly found in the liver and lung but can be discovered in nearly every organ [4]. Surgical methods for dealing with pulmonary cysts include enucleation of intact cysts, and cystotomy, with or without capitonnage, for complicated or intact cysts. The impact of capitonnage on surgical outcome is unknown, and the technique continues to be performed at the choice of the surgeon. The aim of this study was to evaluate the effect of capitonnage in operation for pulmonary cysts. Material and Methods The cases of 75 consecutive patients with pulmonary hydatid disease who were admitted to our clinic and were operated on between January 1994 and September 2001 were retrospectively reviewed. The mean age of the patients was years (range, 5 to 70 years). In all Accepted for publication March 26, Address reprint requests to Dr Turna, Cami Sok. Muminderesi Yolu., Emintas Camlik Sit. No: 32/22, Sahrayicedid, Kadikoy, Istanbul, Turkey; aturna@turk.net. patients, posteroanterior and lateral chest radiography, computed tomography of the chest and upper abdomen, biochemical and hematological testing, and electrocardiography were performed. Fiberoptic bronchoscopy was done if a mass lesion rather than a hydatid cyst was suspected preoperatively. Rigid bronchoscopy was indicated when an endobronchial lesion (eg, of the membrane or endobronchial tumor), was thought to be present. Additional investigations such as the Casoni intradermal test or specific anti-echinococcus immunoglobulin E test were performed in 29 patients who were suspected to have hydatid disease but had no diagnostic findings indicative of this. Arterial blood gas studies were carried out for all patients, and spirometry was performed for those older than 10 years. A diagnosis of pulmonary hydatid cyst was made on the basis of chest radiographic findings, computed tomographic results, or medical history (mainly hydatoptysis [vomitlike expectoration of germinative membrane or hooklets of the parasite]; n 11). Despite all efforts, pulmonary hydatid disease was not diagnosed preoperatively in every patient. Cystic perforation, infection, and calcification and cysts in association with pleural complications in the lung are included in a special clinical entity called complicated hydatid cysts [5]. An infected complicated cyst causes damage to the adjacent lung parenchyma and affects 2002 by The Society of Thoracic Surgeons /02/$22.00 Published by Elsevier Science Inc PII S (02)

2 192 TURNA ET AL Ann Thorac Surg CAPITONNAGE FOR PULMONARY HYDATID CYSTS 2002;74:191 5 wound healing after surgical treatment. However, the term complicated does not necessarily indicate an infected cyst. A perforated cyst without infection is also classified as complicated. Thirty-seven patients had at least one complicated cyst. Of those 37 patients, 5 had a cyst or cysts opened to the pleural space. Operative Techniques A posterolateral thoracotomy in the fifth or sixth intercostal space was accomplished with the patient under general anesthesia and in the lateral decubitus position. Most patients had a double-lumen endotracheal tube. After the cyst was identified, it was surrounded by 1% (vol/vol) povidone-iodine impregnated gauze to prevent seeding of possible daughter cysts. Intact cysts were enucleated in all instances without needle aspiration. However, the cysts were unintentionally ruptured in 3 of the 34 patients with intact cysts. In the 37 patients with complicated cysts, after removal of remnants of germinative membranes, the residual cavity was carefully cleaned and reexamined for spillage from daughter vesicles. In all patients, the pericystic cavities were irrigated with 1% povidone-iodine solution. Bronchial openings were found using saline solution and closed with 3-0 polyglactin 910 (Vicryl; Ethicon, Edinburgh, Scotland). No pericystotomy was performed. However, in complicated cysts with a calcified or thickened pericystic layer, management of bronchial openings was done more carefully with closer and deeper sutures. Five patients underwent a lobectomy (two left upper lobes, two left lower lobes, and one middle lobe) because the cyst involved more than half the lobe. In 4 of these 5 patients, no procedure other than resection was performed, and they were excluded from the study. Thus the results in 71 of the 75 patients were analyzed. Four surgeons did all the operations. Two of them performed capitonnage after closure of bronchial openings during the study period, whereas the other 2 did not. Depending on the preference of the surgeon and independent of the intraoperative findings, all the cavities were obliterated by imbricating sutures from within (capitonnage) using the same suture material in 39 patients (group 1). In 32 patients (group 2), the cavities were left open after closure of bronchial openings. Four wedge resections, which were additional to the cavitary procedures (ie, capitonnage or only closure of bronchial openings), were performed because of damage to the adjacent parenchyma of the involved lobe caused by ruptured or infected cysts, or both. With application of positive intrapulmonary pressure, air escaping through any bronchial opening was visualized by the formation of bubbles. This maneuver was repeated in each patient until all air leaks were sealed. In 11 patients, the diaphragm was opened to treat additional hepatic (n 8) or splenic (n 3) cysts. A splenectomy was performed in 1 patient. In other patients with hepatic or splenic cysts, cystotomy and evacuation of the cystic cavity followed by irrigation with 1% povidone-iodine solution were done. In each patient, 32F or 28F chest tubes were positioned posteriorly and anteriorly. All of the patients were transferred to the intensive care unit after the operation. During the postoperative period, chest tubes were placed on 20 to 25 cm H 2 O suction and were removed when no air leak was evident and when the drainage was less than 150 ml in 24 hours. As prophylactic treatment, cefuroxime sodium, 1.5 g intravenously, administered 1 hour before the operation, was continued every 12 hours for 48 hours postoperatively. In patients younger than 10 years, the dose was reduced to 750 mg twice a day. In 1 patient with a possible nontreated very small cyst ( 1 cm) (ie, nondetectable at operation), albendazole treatment, 400 mg/day, was given for 6 months. The diagnosis of empyema was made when the thoracic drainage became turbid and the presence of bacteria was noted in the fluid. After discharge, the follow-up regimen consisted of physical examination with chest radiography every 6 months. Computed tomography of the chest was indicated when a suspicious lesion appeared on a radiograph. The impact of capitonnage on surgical results was assessed by total hospitalization time, stay in the intensive care unit, duration of air leak from the chest tubes, occurrence of empyema, recurrence, and prolonged air leak ( 7 days). Statistical Analysis Data are reported as the mean the standard error of mean or as percentages. SPSS software for Power Macintosh computers was used on a personal Apple computer for the statistical analysis. Student s t test for paired data was employed for quantitative variables and either the 2 test or Fisher s test, for qualitative variables. A p value of less than 0.05 was considered significant. Results Clinical findings, chest radiographs, thoracic computed tomographic scans, and Echinococcus serologic studies permitted the correct preoperative diagnosis of pulmonary hydatid disease in 49 patients (69%). In the remaining 22 patients, the diagnosis was made intraoperatively. The mean number of cysts per patient detected by computed tomography was (range, one to ten). The total number of treated cysts was 103 in 71 patients. The diameter of the cysts as calculated using computed tomography ranged between 1 cm and 15 cm. Sixteen patients had at least one cyst larger than 10 cm in diameter. Five patients, of whom 1 had bilateral pulmonary cysts, had simultaneous liver hydatid disease. There were 5 patients with bilateral pulmonary cysts. In the preoperative evaluation, specific anti-echinococcus immunoglobulin E test results were positive in 21 (72.4%) of the 29 patients so examined. Characteristics of the patients and locations of the lung cysts are shown in Table 1 and Table 2, respectively. In the preoperative evaluation, cysts were found in the lower lobes in 35 patients. The two groups did not differ in regard to sex, forced expiratory volume

3 Ann Thorac Surg TURNA ET AL 2002;74:191 5 CAPITONNAGE FOR PULMONARY HYDATID CYSTS 193 Table 1. Patient Characteristics a c Variable Group 1 Group 2 Sex (M/F) 21/18 20/12 Age (y) 25.8 (17.6) 34.3 (15.9) FEV 1 (% predicted) 80.4 (23.3) 81.1 (26.1) Cyst diameter (cm) 6.2 (3.6) 6.7 (3.9) No. of cysts 1.38 (0.67) 1.5 (1.6) No. of intact/complicated cysts 20/19 14/18 a Where applicable, data are shown as the mean with the standard deviation in parentheses. b There were no significant differences between the two groups. c Group 1 underwent capitonnage; group 2 had only closure of bronchial openings. FEV 1 forced expiratory volume in the fist second. in the first second, number of cysts, cyst diameter, and number of intact and complicated cysts (see Table 1). There were no intraoperative deaths, and no patient died during the 30-day postoperative period. In 2 patients, additional intrapleural cysts were discovered and resected. In the 5 patients with bilateral disease, bilateral two-staged thoracotomies were performed. At a mean follow-up of 22 months, three recurrences had been detected in group 1 only ( p 0.19). All patients with recurrence had had intact cysts and were treated again through the same thoracotomy. Enucleation and capitonnage had been performed in these 3 patients. The repeat operations were not included in the analysis. Surgical technique had no significant effect on postoperative variables such as presence of empyema ( p 0.47) duration of air leak ( p 0.87), stay in the intensive care unit ( p 0.90), total hospitalization time ( p 0.91), and occurrence of prolonged air leak ( p 0.89) (Table 3). In terms of postoperative radiology, there was no difference between patients who did and did not have capitonnage. Figures 1 and 2 show the preoperative and postoperative posteroanterior radiographs of a patient from each group who had cysts in the left lower lobe. Albendazole (400 mg/day) was administered for 6 months in 4 patients in group 1 and 3 in group 2 with multiple cysts. In 2 of the 3 patients with recurrence, unintentional rupture of the intact cyst had occurred during operation. One recurrence involved a patient with multiple complicated cysts. On the other hand, 1 patient in whom there was spillage of cyst fluid during the operation did not have development of recurrent hydatid disease. Table 2. Locations of Lung Cysts a Location Group 1 Group 2 Right lung Upper lobe 7 5 Middle lobe 6 5 Lower lobe 9 9 Left lung Upper lobe 7 6 Lower lobe 11 9 a Group 1 underwent capitonnage; group 2 had only closure of bronchial openings. Table 3. Relationship Between Surgical Technique and Postoperative Variables a c Variable Comment Group 1 (n 39) Group 2 (n 32) p Value Total hospitalization time (days) Stay in ICU (days) Duration of air leak (days) Empyema Recurrence Prolonged air leak ( 7 days) a Data are shown as the mean the standard error of the mean or as the number of patients affected. b There were no significant differences between the two groups. c Group 1 underwent capitonnage; group 2 had only closure of bronchial openings. ICU intensive care unit. Although some reports [6, 7], suggest medical therapy with albendazole surgical intervention has been the state-of-the-art therapy for pulmonary hydatid disease. The current treatment of hydatid disease of the lung is complete excision of the disease process with maximum preservation of lung tissue. Regarding the management of the cystic cavity in complicated or intact cysts, Délbét [8] first described the method called capitonnage, which is the folding of the pericystic zone by sutures. Crausaz [5] reported that pursestring sutures from the base of the pericystic cavity upward could be used to obliterate the cavity. It is generally agreed, however, that the most important point in the management of the residual pericystic cavity is closure of patent bronchial openings. In both of our study groups, surgical intervention primarily consisted of closing all bronchial openings after removal of the membrane of the cyst. A different approach involves the wall of the pericystic cavity. We concluded that capitonnage did not provide an advantage in hospitalization time, intensive care unit stay, duration of air leak and chest tubes, or prevention of complications such as empyema, recurrence, or prolonged air leak. Saidi [9] pointed out that approximating and suturing cavity edges is not necessary because the pulmonary parenchyma obliterates the space, and the surface of the lung at the site of the residual cavity is covered by pleura. Capitonnage provides complete obliteration of the pericystic cavity to prevent air leak from residual bronchial openings. Without capitonnage, the wall of the pericystic cavity is supposed to be covered by epithelial cells for an uncertain length of time. Experimental studies are needed to test this hypothetical possibility. Similarly, in a pediatric series, Çelik and associates [10] showed no advantage for capitonnage in operations for pulmonary hydatidosis, and therefore they did not advocate the technique. They also found that capitonnage could cause atelectasis by obliterating the bronchus surrounding the cyst. We observed no such complication in one group 1 patients. Moreover, postoperative chest

4 194 TURNA ET AL Ann Thorac Surg CAPITONNAGE FOR PULMONARY HYDATID CYSTS 2002;74:191 5 stated that, enucleation of larger cysts carries an increased possibility of rupture during the separation of the pericystic zone from the laminated membrane. Although they used mainly needle aspiration for cystic fluid during operation, Halezaroğlu and associates [14] recommended enucleation rather than needle aspiration whenever the risks did not overweigh the advantages. Taking into account the recurrences in our study, one could recommend needle aspiration of the cysts instead of enucleation as the procedure of choice for intact cysts. Although pericystotomy (ie, Perez-Fontana operation Fig 1. Posteroanterior chest radiographs of a patient with left lower lobe cyst (A) before and (B) after surgical treatment with capitonnage. radiographs showed no significant difference between patients with and without capitonnage. Recently, Sonmez and associates [11] reported that capitonnage shortened the postoperative chest tube drainage period and reduced morbidity compared with no capitonnage, but the report involved a pediatric population of 15 patients. In our study, all three recurrences were in group 1, but the difference did not reach significance. Concerning this finding, there might be a potential bias attributable to the enucleation process itself because 2 (66.7%) of the 3 patients in whom accidental rupture of intact cysts occurred during the operation had recurrence. Enucleation is the preferred method in our institution but is not favored by others [10, 12, 13]. Symbas and Aletras [13] Fig 2. Posteroanterior chest radiographs of patient with left lower lobe cyst (A) before and (B) after surgical treatment without capitonnage.

5 Ann Thorac Surg TURNA ET AL 2002;74:191 5 CAPITONNAGE FOR PULMONARY HYDATID CYSTS 195 [15]) is carried out in most series [3, 9, 11, 13], we did not choose to perform this technique because it prolongs the operation time and increases the risk of hemorrhage. In addition, we believe that if the cystic cavity is properly obliterated, pericystic tissue causes no complication. Despite the fact that we did not use that method in our series, our complication rates were comparable to those of others [3, 4, 10, 12, 14]. Pulmonary resection must be avoided as much as possible. However, segmental resection, wedge resection, and lobectomy are justified when the size and the number of cysts and the degree of infection exclude lesser procedures [13]. The principal indications for lobectomy are large cysts involving more than 50% of the lobe, cysts with severe pulmonary suppuration not responding to treatment, multiple unilobar cysts, and sequelae of hydatid disease such as bronchiectasis, pulmonary fibrosis, or severe hemorrhage, as capitonnage or any other procedure leads to complications in such patients [13]. In our study, three lobectomies were performed because more than half the lobe was involved, one lobectomy was done for a fibrotic lobe that would not reexpand, and one, for a congested and infected lobe after the first operation (as a second operation). However, 4 of the patients who underwent lobectomy only were not included in the analysis because our aim was to evaluate the effect of capitonnage on surgical outcome. Although pulmonary hydatidosis has not been recognized as an emergency state, anaphylactic shock and subsequent suffocation and death are possibilities [13]. One patient with an intact cyst who was not included in the study died of anaphylactic shock during hospitalization. For this reason, priority on the operation list should be given to patients with an intact pulmonary cyst. This study was not a randomized trial, but the preoperative variables were well balanced between the two study groups. A bias in the surgeons methodological concept is a possibility, and the patients in group 2, were slightly older than those in group 1. These potential biases could be eliminated with a randomized study. The impact of capitonnage on recurrence of hydatid disease was found to be marginal. However, there is a chance that the number of patients was suboptimal to show the effect of the technique. In summary, our study suggests that each of the surgical methods in the hands of its proponents yielded good results. Capitonnage had no beneficial effect on the surgical management of pulmonary hydatid cysts in terms of postoperative variables and short- and longterm surgical complications. Management of the bronchial openings is of major importance, whereas capitonnage can be omitted to shorten the operating time. Further randomized studies are needed to clarify the role of capitonnage in operations for pulmonary hydatid cysts. References 1. Ayuso LA, de Peralta G, Lazaro RB, Stein AJ, Sanchez JA, Aymerich DF. Surgical treatment of pulmonary hydatidosis. J Thorac Cardiovasc Surg 1981;82: Novick RJ, Tchervenkov CI, Wilson JA, Munro DD, Mulder DS. Surgery for thoracic hydatid disease: a North American experience. Ann Thorac Surg 1987;43: Doğan R, Yuksel M, Çetin G, et al. Surgical treatment of hydatid cysts of the lung: report of 1055 patients. Thorax 1989;44: Aytac A, Yurdakul Y, Ikizler C, Olga R, Saylam A. Pulmonary hydatid disease: report of 100 patients. Ann Thorac Surg 1977;23: Crausaz PH. Surgical treatment of the hydatid cyst of the lung and hydatid disease of the liver with intrathoracic evolution. J Thorac Cardiovasc Surg 1967;53: Nahmias J, Goldsmith R, Soibeteman M, el-on J. Three- to 7-year follow-up after albendazole treatment of 68 patients with cystic echinococcosis (hydatid disease). Ann Trop Med Parasitol 1994;88: Mawhorter S, Temeck B, Chang R, Pass H, Nash T. Nonsurgical therapy for pulmonary hydatid cyst disease. Chest 1997; 112: Délbét P. Kystes hydatiques du foie traités par le capitonnage et la suture sans drainage. Bull Mem Soc Chir Paris 1899;25: Saidi F. Surgery of hydatid disease. Philadelphia: WB Saunders, 1976: Çelik M, Şenol C, Keles M, et al. Surgical treatment of pulmonary hydatid disease in children: report of 122 cases. J Pediatr Surg 2000;35: Sonmez K, Turkeyılmaz K, Demirogullari B, et al. Hydatid cysts of the lung in childhood: is capitonnage advantageous? Ann Thorac Cardiovasc Surg 2001;7: Yalçınkaya I, Er M, Özbay B, Uğras S. Surgical treatment of hydatid cyst of the lung: review of 30 cases. Eur Respir J 1999; 13: Symbas PN, Aletras H. Hydatid disease of the lung. In: Shields WT, ed. General thoracic surgery. 5th ed. Philadelphia: Williams & Wilkins, 2000: Halezeroğlu S, Çelik M, Uysal A, Şenol C, Keleş M, Arman B. Giant hydatid cysts of the lung. J Thorac Cardiovasc Surg 1997;113: Perez-Fontana V. Traitement chirurgical du kyste hydatique dus poumon. La methode uruguayenne ou extirpation du perikyste. Arch Int Hydatid 1951;12:469.