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Eur J Cardiothorac Surg 1999;16:628-635
© 1999 Elsevier Science NL
Department of Thoracic and Cardiovascular Surgery, Puerta de Hierro Clinic (Clínica Puerta de Hierro), Autonomous University of Madrid (Universidad Autónoma de Madrid), Madrid, Spain
Corresponding author. Avenida de Valladolid, 57-59, portal 2, 5-B, 28008 Madrid, Spain. Tel.: +34-91-316-2240; fax: +34-91-373-7667
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Abstract |
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 Top Abstract 1. Introduction2. Patients and methods3. Results4. DiscussionAppendix AReferences |
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Key Words: Pulmonary hydatidosis • Surgery • Suction-trocar
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1. Introduction |
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TopAbstract1. Introduction2. Patients and methods3. Results4. DiscussionAppendix AReferences |
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Echinococcosis is endemic to the Mediterranean region, South America, Australia, New Zealand, the Middle East, Alaska and Canada, where it is widespread among Indian tribes [1–7]. In its adult stage, the parasite lives in the intestinal tract of carnivores. The head is composed of a double crown of hook-like structures, and the body is formed by three or four rings, the last of which bears the eggs. After being eliminated with the feces, the eggs contaminate fields, irrigated land and wells. Herbivores ingest the eggs, which develop into larvae, or hydatids, within the viscera of these animals. The cycle is completed with the ingestion of the infected viscera by carnivores. Humans contract the disease from water or food or by direct contact with dogs. Once the eggs reach the stomach, the hexacanth embryos are released. They pass through the intestinal wall and reach the tributary veins of the liver where they undergo a vesicular transformation and develop into the hydatid. If they overcome the hepatic obstacle, they may become lodged in the lung, where they would also be transformed into hydatids. If they advance beyond the lung, they may remain in any organ to which they are carried by the bloodstream. It has been shown that the embryos can reach the lung via the lymphatic vessels, bypassing the liver, and there is also evidence that the disease can be contracted through the bronchi.
Although Felton reported hydatid cyst to be a rare cause of pulmonary nodule, we have found it to be one of the most frequent [6,8].
In Spain, the epidemiological data on hydatid disease have improved considerably and, at the present time, the number of cases reported is minimal, as illustrated by the fact that, in 1966, surgical treatment for this disease was performed in 4.62 patients per 100 000 population per year, while in 1988, the incidence had dropped to 1.77 patients per 100 000 population per year, and in 1997 was recorded at 0.78 patients per 100 000 population per year. The number of hospital admissions for hydatid disease each year has decreased over the course of time from 6690 in 1966, to 2343 in 1988, and down to 986 in 1997. These data were obtained from the Boletín Epidemiológico (Epidemiological Bulletin) which bases its reports on the Survey of the Obligatory Declaration of Infectious Diseases and the Survey on Hospital Morbidity carried out by the Spanish National Statistical Institute.
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2. Patients and methods |
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TopAbstract1. Introduction2. Patients and methods3. Results4. DiscussionAppendix AReferences |
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Twenty-four patients (10%) were asymptomatic and the cyst was discovered incidentally upon plain chest X-ray for other causes. The remainder of the patients presented one or more of the symptoms appearing in Table 1. The diagnosis was confirmed on the basis of additional studies including Casoni's skin test, serological analysis, immunoelectrophoresis, blood eosinophil count (>3%) and erythrocyte sedimentation rate (>20 mm in the first hour).
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All the patients underwent posterolateral thoracotomy. More recently, in cases of multiple and bilateral hydatid cysts, we have utilized bilateral ‘clamshell’ thoracosternotomy.
We employed a trocar-suction device for needle aspiration (Fig. 1). The use of this instrument prevents the rupture of the cyst, eradicates the parasite and makes it possible to excise the residual cavity. The remaining 24 cysts were totally removed using different techniques. To deal with the residual cavity, resection of the pericystic layer and total eversion of the cavity was performed in 86.2% of cases and capitonnage in the remainder. High vacuum suction (-30 cm H2O) was employed in every case (Table 2).
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In patients presenting hydatid cysts at other sites, excision of the ‘other’ cyst was carried out in a second surgical procedure. The decision as to which cyst should be removed first was based on the susceptibility of each to rupture, their size, the risk of dissemination and the vital importance of the organ in which each was located. In cysts of lower right lung lobe reaching the liver through the diaphragm (four cases in this series), the surgical approach (thoracotomy or laparotomy) was chosen according to the size of the cyst and the existence of signs of possible complications. When they appeared to affect the lung, thoracotomy was performed and if the liver was at risk, laparotomy was attempted, although in one such case, the approach was extended to include thoracotomy because of the need to perform right lower lobectomy.
We employed mebendazole at the doses recommended by Bekhti to treat hydatid disease of the lung (400–600 mg every 8 h) as follows [9]. Administration commenced 7–10 days prior to surgery. When the cyst was intact and there appeared to be no risk of its rupture during the surgical procedure, the treatment was administered for 1 month. When the cyst was ruptured or the possibility of its intraoperative dissemination was suspected, treatment was continued for 3 or 6 months. When dissemination was confirmed and total or partial resection of the lesions was not feasible, it was necessary to excise as many of the cysts as possible, attempting to conserve a maximum of lung parenchyma, and administer mebendazole until there was no radiological evidence of the lesions.
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3. Results |
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TopAbstract1. Introduction2. Patients and methods3. Results4. DiscussionAppendix AReferences |
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There were signs of cyst rupture in 70 patients (29%); they consisted of the meniscus sign, an incarcerated membrane (Fig. 4a), the water-lily sign (Fig. 4b), air fluid levels or the double-arch of Ivanissevich. In 20 patients (28.6%), the lesion was an empty cavity and in 34 (48.6%), there were no clear-cut margins defining the lesion. In 8 (11.4%), the cyst was associated with pleural effusion; and in 3 (4.3%), it was calcified.
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Pneumothorax occurred in 24 cases (8.8%) and was resolved by placing chest drainage in 23 cases; in the remaining case, tension pneumothorax developed due to dehiscence of the bronchial suture, followed by irreversible cardiac arrest. Pleural empyema occurred in 10 cases, 9 of which were managed by chest drainage; the remaining patient required another operation to treat an associated bronchopleural-cutaneous fistula. Surgical wound infection developed in 18 cases. It was superficial in 14 and was successfully managed with local treatment. The remaining 4 patients all presented bronchopleural-cutaneous fistula, one of which was resolved by conservative treatment; but the other three required surgical treatment. Details concerning these and other complications that arose during the early postoperative period are shown in Table 3.
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4. Discussion |
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TopAbstract1. Introduction2. Patients and methods3. Results4. DiscussionAppendix AReferences |
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In our series, we assessed some of the diagnostic tests for hydatid disease, such as the eosinophil count, the Casoni test, the Weinberg test and erythrocyte sedimentation rate to determine whether any of them showed a statistically significant correlation. We found none when the cyst remained intact during the preoperative evaluation or the surgical procedure. However, when rupture occurred, an eosinophil level of over 3%, episodes of vomica, expectoration of ‘spring water’ and small particles of hydatid cyst membrane with the aspect of grape skin, and the existence of three or more clinical symptoms showed statistically significant correlations. Ales et al. employed indirect immunofluorescence with better results than the preceding studies [2,12].
Bronchial endoscopy has been utilized in the diagnosis of pulmonary hydatidosis with considerable success prior to ultrasound and other imaging techniques such as CT and MRI; its use has been limited by the risk of rupture of the cyst and the development of severe complications. It may be indicated in cases of ruptured hydatid cyst of the lung since it enables the visualization of the hydatid membrane, resembling ‘the shininess of cooked egg white’, and small fragments of the cystic membrane can even be removed during bronchoscopic examination using the technique of Coll and Collomé [13].
Diagnostic bronchography, the approach employed years ago, has followed the steps of bronchoscopy, being replaced by imaging techniques such as CT and MRI [5,6,14].
In clinical practice, plain chest X-ray is the study that has shown to be most reliable in the diagnosis of pulmonary hydatid disease. Numerous images have been described, ranging from those typical of the intact cyst, of round or oval shape, solitary or multiple, of homogeneous density and having a perfectly defined margin as though it were drawn with a marking pen, to ruptured or complicated cysts with the membrane floating in fluid resembling the water-lily sign; an incarcerated membrane folded back in the form of a barricade or the Moral Torres sign which appears as a fluid-filled abscess, with or without air-fluid levels; images of pneumonitis of the lung; or areas of lung atelectasis. In these cases, the differential diagnosis with certain lesions such as bronchial tumors, sarcomas or tuberculomas is a difficult challenge [6].
The incision employed was lateral thoracotomy in cases of solitary pulmonary cysts. In the earlier cases, when bilateral cysts were present, the operation was done in two stages, with a first thoracotomy on the side on which the risk of rupture appeared to be greater or the cyst was more complicated [15]. We now treat these cases in a single procedure using a median sternotomy for cysts located in anterior position and transsternal bilateral thoractomy for cysts in anterior or posterior position or associated with hydatid disease of the heart. We have also performed anterior bilateral thoracotomy in a single procedure in some cases.
The objective in the surgical treatment of pulmonary hydatidosis is to eradicate the parasite, to prevent the intraoperative rupture of the cyst with its subsequent dissemination and to remove the residual cavity. Most authors agree that the 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 [6].
Initially, the surgical treatment of pulmonary hydatidosis involved the marsupialization of the cyst when it was attached to the wall, or an atypical pulmonary resection consisting of two stages: first pleurodesis was produced, followed by marsupialization in a second procedure. Evidently, these techniques have since been abandoned.
Simple cystectomy with capitonnage or approximation of the pericystic tissue (the method of Posadas), or cystopericystectomy (the Pérez Fontana method) have also been carried out. Whenever possible, ‘when the cyst is small and there is no risk of rupture’, its complete removal can be attempted, aided by an increase in the airway pressure provided by the anesthetist (the Ugón technique).
In our series, we used needle aspiration involving the trocar-suction device designed by Professor D. Figuera in 276 (92%) of the cysts we treated surgically [16]. With this approach, we have observed no recurrences of pulmonary hydatidosis in those cases in which the cysts remained intact at the time of the operation [6]. This device comprises a trocar containing a needle connected to a system of negative pressure aspiration and surrounded by a suction cup that fits over the convex part of the cyst wall. When the device is applied to the cyst, the negative pressure makes the suction cup adhere hermetically to the cyst wall, thus impeding the extravasation of the content as it is sucked out, and eliminating the risk of intraoperative contamination. This surgical technique has been shown to be effective in preventing the rupture and dissemination of the cyst and facilitates the excision of the residual cavity.
In patients with hydatid cysts at other sites, we treat the ‘other’ cyst in a second surgical procedure. The decision as to which cyst should be removed first was based on the susceptibility of each to rupture, their size, the risk of dissemination and the vital importance of the organ in which each was located [17,18].
In four cases, cysts in lower right lung lobe were connected to hepatic cysts which passed through the diaphragm, resembling an ‘hour glass’ [19–22]. The surgical approach to these lesions is a matter of discussion. We chose thoracotomy or laparotomy, depending on the size of the cyst and the existence of signs of complications. When they appeared to affect the lung, thoracotomy was performed, and if the liver was at risk, laparotomy was attempted, although in one such case, the approach was extended to include thoracotomy because of the need to perform right lower lobectomy. Some of the authors who propose the systematic performance of thoracotomy have had to extend the incision to laparotomy for T-tube placement because the hepatic cyst involved the bile duct.
We performed atypical resection when rupture or peripheral infection was detected. Pulmonary lobectomy was carried out when the cyst was associated with abscesses or severe pulmonary changes or with atelectasis that remained unresolved by standard surgical procedures, as well as when there was evidence that a given lobe contained infected hydatid membranes or several cysts. All authors agree on the need to respect as much of the lung tissue as possible, performing resection only when the lung is destroyed [6,23–25].
Whenever possible, we opt for complete resection of the pericystic tissue with total eversion of the cavity. We performed capitonnage in only 13.7% of our patients since we attribute the persistence of residual cavities to this measure. The surgical area was protected with aspiration and compresses soaked in formaldehyde during the early stage of the series to prevent the dissemination of the contents. The formaldehyde was later replaced by hypertonic saline solution to avoid the inflammatory reaction produced by formaldehyde. The pleural space was aspirated with elevated negative pressure (-30 cm H2O) in every case.
Since the first pilot studies with mebendazole in 1974, we have employed this drug as adjuvant therapy according to the protocol proposed by Bekhti (400–600 mg/8 h) as follows [9]. Administration commences 7–10 days prior to surgery. When the cyst is intact and there appears to be no risk of its rupture during the surgical procedure, the treatment is administered for 1 month. When the cyst is ruptured or the possibility of its intraoperative dissemination is suspected, treatment is continued for 3–6 months. When dissemination is confirmed and total or partial resection of the lesions in not feasible, it is necessary to excise as many of the cysts as possible, attempting to conserve a maximum of lung parenchyma, and administer mebendazole until there is no radiological evidence of the lesions. In our patients, we have observed the same macroscopic and microscopic changes reported by other authors. We have since substituted albeldazole for mebendazole, using the same regimen.
We consider that the excellent results observed in this series, with a survival rate of 94.6% after 18 years of follow-up, 95.1% of patients free of hydatid disease and 98.3% free of lung disease, demonstrates the long-term efficacy and safety of the trocar suction device employed here for needle aspiration of pulmonary hydatid cysts.
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Acknowledgments |
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Footnotes |
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Appendix A |
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TopAbstract1. Introduction2. Patients and methods3. Results4. DiscussionAppendix AReferences |
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Dr Varela: Yes, we protect the field with hypertonic saline-soaked sponges. Now we use the double lumen tube, and in noncomplicated cases we inflate the lung because it facilitates the enucleation. But if you should have any doubt that the cyst is in direct contact with the bronchial airway, you ought to clamp the bronchus.
Dr S. Mattila (Helsinki, Finland): We have seen quite a few cases in Finland. The vector animals in those cases have been reindeer and dog. Earlier enucleation has been the treatment. But also in the latest cases we have used the aspiration technique and injection of alcohol to the cavity. Such a treatment has been used also in liver hydatid cysts.
Dr T. Dosios (Athens, Greece): What I understand is that before you apply aspiration, you open the capsule, do you?
Dr Varela: Never.
Dr Dosios: How does the cyst come out of the capsule with this aspiration only? There is no rupture?
Dr Varela: You just aspirate the contents, not the membrane.
Dr Dosios: So the membrane remains inside the lung?
Dr Varela: The cyst contents are aspirated.
Dr Dosios: Yes.
Dr Varela: But the membrane is embedded in the lung, so it cannot be aspirated. Then you can do a partial resection or total resection of the pericyst membrane, if it is possible, or just to capitonnage if no leaks are observed.
Dr Dosios: What about the infected cysts; what is the percentage of infected cysts you have in your series?
Dr Varela: All of them were aspirated with this vacuum device.
Mr J. Khalil Marzouk (West Midlands, UK): Your series dates back to 1966. When exactly was the device introduced and did you see any pleural disease, was there any difference between the series before the device and after?
Dr Varela: The device was introduced in 1953 by Dr Figuera, but had previously been used for treating lung abcess or even empyemas, and thereafter, it was used for hydatid cysts. When you have a dissemination to the pleura or to the bronchial airway, you have a problem, and sometimes you have dissemination to both lungs, and pleura. In those cases patients must be treated almost forever medically.
Dr A. Tcherveniakov (Sofia, Bulgaria): I only wanted to mention that actually the sheep are just the intermediate recipient and the dogs are the real threat for people. I would like to ask you about your experience with bilateral hydatid cysts of the lungs. What tactics and approach do you use to treat them, because in our practice for recent years the number of bilateral cysts of the lungs treated on one stage has increased?
Dr Varela: Yes. As you know, the gold standard for hydatid cyst is always surgery, and in the last year we used a bilateral anterior thoracotomy, cross-cutting the sternum. We think this approach is very useful; you can explore both lungs and you can use this technique for both lungs.
Dr M. Amin (Cairo, Egypt): If you have hydatid cyst at multiple sites, do you start with surgery or with intervention? For example, if you have one or both sites on the lungs and one in the liver, which part do you start with?
Dr Varela: I think it is a very good question because sometimes we have seen that kind of patient. I think you have to individualize each patient. If the problem is in the liver with a big, huge hydatid cyst, I think you should begin with the liver, but if you have multiple different sites in both lungs, I would advise to start with bilateral anterior thoracotomy and then the liver. I do not think there is a rule for that very special patient.
Dr A. Nasri (Zalaegerszeg, Hungary): Sometimes we used to see the hydatid cysts combined with small satellite cysts. So have you noticed this thing, and if you noticed it, how can you treat these satellite cysts?
Dr Varela: Yes. It depends on the presence of atelectasis. I mean, if during the operation and after the resection of the cyst the lung may be inflated and microscopically you think that it is all right, you just leave it, but many times the atelectasis is inflammatory atelectasis the lung cannot be inflated, so in that case I think it is better to resect that part of lung.
Dr Nasri: Do you treat the patient with medication after the operation or not?
Dr Varela: Yes. As you know, we have guidelines for this disease, and in any case we treat at least one month after surgery with mebendazole or albendazole. But in complicated cases like rupture of the cyst, you should treat these patients for more than three or six months.
Dr O. Oto (Izmir, Turkey): I have got two comments on double lung bilateral hydatid cyst and concomitant liver and lung disease. For the bilateral hydatid cyst our standard approach is median sternotomy. Through median sternotomy you can even reach to the back side of lung and you can remove it easily. As it is well known, this incision is less painful. Secondly, if we have the cyst at the apex of the liver and at the right side of the lung, we extract the cyst of the liver through the diaphragm at the same time simultaneously.
Dr Varela: Well, I think there are many ways to treat these patients but we are very comfortable with the anterior bilateral thoracotomy, which we use for lung transplantation. The post op of these patients is similar to the sternotomy, and for this kind of disease, in the left lower lobe, we feel more comfortable with this approach. But you have a good experience in your country with a lot of patients and maybe a median sternotomy is enough.
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References |
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TopAbstract1. Introduction2. Patients and methods3. Results4. DiscussionAppendix AReferences |
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