Abstract
Background
Multiple hydatid disease is a complex surgical problem, and its treatment can follow either conservative principles (drainage or obliteration of the cavity) or radical principles (cystoperi-cystectomy or liver or lung resection).
Methods
A total of 220 patients with multiple cystic echinococcosis (428 cysts) were managed between 1967 and 1998 with conservative operations (group A) or radical operation (group B). There were 90 men and 130 women, with a mean age of 52 years (range 18–77 years). There were two cysts in 124 patients, three cysts in 40 patients, four in 15 and more than four in 41 patients. These multiple cysts were located at one anatomical site (n=140) or at more than one site (n=80). Multiple (2–3) hepatic cysts occurred in 142 patients, multiple (2–3) lung cysts in 15 and multiple peritoneal cysts in 13 patients. Hepatic cysts co-existed with lung cysts in another 32 patients, with peritoneal cysts in 14 patients and once each with splenic, splenic plus lung cysts and renal cysts, one retroperitoneal cyst coincided with small peritoneal cysts.
Results
The operative procedure employed was dependent on the type and site of the parasite and the condition of the host. Three of 208 patients operated conservatively (group A) died postoperatively as opposed to receiving radical treatment. Morbidity rates were 8.8% and 12.5% in group A and B and mean hospital stay was 15.8 and 15.1 days, respectively. In group A there was an 8.6% recurrence rate, and recurrent disease was finally managed in each group the overall result could be considered satisfactory.
Discussion
We conclude that conservative surgery can provide good results in multiple cystic echinococcosis. Radical surgery, with its time-consuming major procedures, is ideal but only in properly selected cases.
Keywords: multiple hydatid disease, cystic echinococcosis
Introduction
Besides the known complications of hydatid disease (rupture, suppuration and calcification), multiplicity of cysts is another common surgical problem 1,2,3. Sometimes the management of multiple cysts is difficult, requiring sound operative experience, preferably with a one-stage procedure and after appropriate preoperative preparations. In this study we report our experience with this severe echinococcal infestation, arising in one or more locations in the body, with the specific aim of comparing the results of conservative and radical surgical approaches.
Materials and Methods
A total of 615 patients received operation for hydatid disease at this clinic between 1967 and 1998, and of these nearly one-third (n=220) had involvement of more than one organ. Multiple cysts occurred in one organ (n=140) or more than one organ (n=80). There were 90 men and 130 women with an age range from 18 to 77 years (mean 52) (Table 1). Careful preoperative screening revealed co-existent diseases in 39 patients. The 220 patients operated for multiple cysts had a total of 428 cysts: two cysts in 124 patients, three in 40, four in 15 and more than four in 41 patients. The clinical picture was dependent on an expanding cyst, the development of complications and localisation of the parasite (Table 2). The diagnosis was also based on various serological and isotopic tests (early cases), but mainly on sonographic and radiological examinations in more recent cases (Figures 1 and 2). The incision and operative planning depended on the location and type of the parasite and the patient's general condition, operations being performed in one stage (202 cases) or two stages (7 cases). In cases of co-existing abdominal and lung cysts, the pulmonary cysts were operated first, except for cysts of the right lung and dome of the liver which were operated simultaneously. Bilateral lung cysts were resected in two stages, with a 2–4-week interval. The lung with larger cysts, multiple cysts or uncomplicated cysts was operated first.
Table 1. Location of multiple hydatid cysts.
| Location | n |
|---|---|
| Liver | 142 |
| Lung | 15 |
| Peritoneum | 13 |
| Liver + lung | 32 |
| Liver + peritoneum | 14 |
| Liver + spleen | 1 |
| Liver + lung + spleen | 1 |
| Liver + kidney | 1 |
| Retroperitoneum + peritoneum | 1 |
| Total | 220 |
Table 2. Cyst characteristics in multiple hydatid disease cases.
| Parameter | n |
|---|---|
| Multivesicular | |
| Liver | 187 |
| Ruptured | |
| - Liver in bile duct | 3 |
| - Liver in bronchial tree | 5 |
| - Lung in pleura | 3 |
| Suppurated (primarily) | |
| - Liver | 16 |
| - Lung | 6 |
| Calcified | |
| - Liver | 25 |
Size of cysts 2–25 cm (mean 13 cm)
Figure 1. .
CT scan: multiple hydatid cysts in the right lobe of the liver and in the left lung.
Figure 2. .
Abdominal CT scan: multiple hydatid cysts in both lobes of the liver and in the spleen.
In most cases a ‘conservative’ surgical approach was chosen (group A: 404 cysts). The main objectives of this treatment were the avoidance of spillage of cyst contents, the removal of all parasitic elements and the management (if possible), of the residual pericyst cavity by various methods. Before the evacuation and necessary partial pericystectomy, the initial steps were decompression of the cysts by needle and trocar, removal of the parasite and, as a scolicidal procedure, lavage of the cavities with hypertonic saline swabs. The opening of the pericyst was sutured, or the cavity was left open, and either obliterated by capitonnage (in folding of the cystic wall by successive suturing layers) or filled with omenturn. In infected cysts or hepatic cysts with intrabiliary rupture, external drainage of the cavities was usually undertaken. In cysts of the liver that had ruptured into the biliary tree, the operative technique was directed towards the cyst, but in addition towards possible suturing of the cystobiliary communication and drainage of the common bile duct (T tube, two cases, choledo-duodenostomy, one case). The gallbladder was excised, if involved (nine cases). In biliobronchial fistula due to thoracic extension of a cyst of the dome of the liver, besides management of the parasitic cavity, suturing of the pulmonary side of the fistula and closure of the diaphragmatic defect was the usual procedure. In calcified cysts of the liver, operated because of their size or co-existent infection, pericystectomy was performed with removal of the calcareous plaques and drainage. In huge calcified cysts a ‘planned’ re-operation was sometimes carried out with a bloodless chiselling of the calcification (due to intervening inflammation). In lung hydatids the two general principles were to remove the parasite and to treat both the bronchipericyst pathology and associated lesions as conservatively as possible. The procedure of choice was the tedious delivery of the parasite by enucleation. Obviously for large cysts, needle aspiration and extraction without spillage of the collapsed cysts were essential. Especially important was the closure of any bronchial openings in the remaining cavities.
In the remaining 24 patients (group B), with enormous cysts or penduculated peripheral cysts, a ‘radical’ approach was adapted-either cystopericystectomy or liver or lung resection was performed (group B = 24).
The procedures performed are listed in detail in Table 3.
Table 3. Operative procedure and site of cysts in multiple hydatid disease.
| H | L | P | H + L | H + P | Other | |
|---|---|---|---|---|---|---|
| Group A (404 cysts) | ||||||
| Partial pericystectomy, capitonnage | 24 | 7 | – | 12 | – | – |
| Partial pericystectomy, omentoplasty | 3 | – | 1 | 1 | 1 | – |
| Partial pericystectomy, drainage | 182 | – | 15 | 25 | 21 | 5 |
| Evacuation via another superficial cyst | 2 | |||||
| Evacuation via CBD | 1 | |||||
| Enucleation, capitonnage, closure of the bronchial openings | – | 24 | – | 28 | – | – |
| Enucleation | – | 15 | – | 16 | – | 1 |
| Enucleation with pleurectomy | – | – | – | 1 | – | – |
| Removal intact pediculated | – | – | 12 | – | 6 | 1 |
| Group B (24 cysts) | ||||||
| Cystopericystectomy | 8 | 1 | – | 8 | 1 | – |
| Lobectomy | – | – | – | 2 | – | – |
| Segmentectomy | – | – | – | 1 | – | – |
| Splenectomy | – | – | – | – | – | 2 |
| Nephrectomy | – | – | – | – | – | 1 |
| Total = 428 cysts |
H, hepatic; L, lung; P, peritoneal; H + L, Hepatic + lung; H + P, hepatic + peritoneal.
Results
The overall postoperative morbidity rate (including re-operations), was 10.6%, i.e. 8.8% in group A and 12.5% in group B. The complications in each groups according to cyst location are listed in Table 4. The mean hospital stay was 15.5 overall days; 15.8 in group A and 15.1 days in group B. Except for total complication in three patients, most of the postoperative complications were easily treated. The re-operations are summarized in Table 5. Three patients in group A died postoperatively.
Table 4. Morbidity and hospital stay in patients operated for multiple hydatid cysts.
| Morbidity (no. of patients) |
Mean hospital stay (days) |
|||
|---|---|---|---|---|
| Group | Group | |||
| Site | A | B | A | B |
| Liver | 12 | 1 | 23 | 18 |
| Lung | 1 | 1 | 18 | 14 |
| Peritoneum | 2 | – | 10 | 8 |
| Liver + lung | – | – | 16 | 12 |
| Liver + peritoneum | 2 | – | 10 | 8 |
| Liver + spleen | 1 | – | – | 14 |
| Liver + lung + spleen | – | – | – | 26 |
| Liver + kidney | – | – | – | 21 |
| Retroperitoneum + peritoneum | – | – | 18 | – |
Table 5. Re-operations for multiple hydatid cysts.
| Group A | |
|---|---|
| *Liver | 12 |
| Lung | 1 |
| Peritoneum | 2 |
| Liver + lung | – |
| Liver + peritoneum | 2 |
| Liver + spleen | 1 |
| Liver + lung + spleen | – |
| Liver + kidney | – |
| Retroperitoneum + peritoneum | – |
| Total | 18 |
*Including eight operations ‘planned’ for chiselling the remaining calcinous layer of the cysts and easy collapse of the cavities.
One patient, with multiple cysts of the liver and chronic obstructive pneumonopathy, died on the fifth postoperative day from heart failure. One patient, with multiple infected cysts of the liver, died on the 146th postoperative day from MOF (sepsis). One patient, with multiple hydatidosis of the liver, died on the 23rd postoperative day from sepsis. During a follow-up period of 1–35 years the recurrence rate was 8.2%. These patients, all in group A, were treated surgically without a death.
Discussion
The choice of operative method in cystic echinococcosis remains controversial 1,2. In patients with multiple cystic involvement, it is even more difficult to decide on the type and timing of management, hence our attempt to compare various techniques with regard to postoperative morbidity, hospital stay and late results 3.
In contrast with other studies, there was a female preponderance and a higher mean age in the present series 4. The 35.8% rate of multiple cysts was higher than that reported elsewhere (18.4–28.8%) 4,5.
Although the spectrum of symptoms is highly varied, the tumour-like signs prevail, with the characteristic features secondary to cyst rupture being very common 4,5,6. The diagnosis of the disease is generally easy 7. Ultrasonography and computed tomography are now widely used, replacing the use of isotopes in most cases. The intradermal Casoni's test no longer has a place. Serological examinations have the problem of low diagnostic sensitivity and specificity and are now mainly used for differential diagnosis of larval cestode infections 8.
We used long incisions for maximal exposure of the cyst and surrounding structures, with the approach of choice dependent on the localisation of the parasite 9. In cysts of the upper portion of the liver, thoracic plus abdominal incision, or rarely a thoracoabdominal incision was mandatory to permit complete evacuation of the cysts without spillage, adequate pericystectomy and the proper management of the remaining cavities.
In accordance with some authors but in contrast to others most of our patients were operated conservatively because of the anatomy and characteristics of the cysts 3,4,7. In uncomplicated liver cysts the therapeutic objectives were the cautious removal of the parasite, and the management of the residual cavity. In cysts that had ruptured in the biliary tree the operative technique was directed at drainage of the common bile duct (besides managing the cyst). In liver echinococcosis with intra-biliary rupture and primary or secondary suppuration, proper drainage was required by means of wide-bore tubes. In huge calcined hepatic cysts, in the initial operation or in the re-operation, debulking of the calcareous wall of the adventitia and easy collapse of the parasitic cavity was necessary.
In some peritoneal cysts, partial pericystectomy with parasite evacuation and drainage was selected because of adhesions around the cysts and the danger of bleeding or rupture of the parasite during the dissection.
A few cases require more radical procedures 1,4. In our series cystopericystectomy was the ideal technique for small calcified hepatic cysts or cysts that were peripherally located or pedunculated, whenever this time-consuming method allowed a plane to be established between the parasite and the host's tissue. Formal resections were rarely performed for liver and lung cysts because these radical procedures seemed disproportionate for a benign lesion and because of the possible risk of re-infection in the remaining healthy part. In two splenic cysts and one renal cyst splenectomy and nephrectomy were carried out because of destruction of these organs.
Some therapeutic principles emerge from these series of multiple cysts 7,9,10. In multiple cases it is better to manage all the cysts in one stage, adjacent parasites being easily evacuated through the cavity of the more superficial cyst. Otherwise, when one of the cysts is in an inaccessible position and the patient's condition is poor, it is best to plan a two-stage procedure. In co-existing liver and pulmonary parasites the lung cysts should be operated either first, because of the greater tendency to rupture and infection, or simultaneously (in cysts of the dome of the liver and right lung). Bilateral lung cysts have to be resected in two stages. In uncomplicated lung cysts the lung with the larger cyst or with more cysts must be treated first. In patients with unruptured cysts >5 cm in one lung and a ruptured cyst in the other, the intact lung has a higher therapeutic priority.
It is difficult to compare our results with those from other centres because few reports have concentrated on multiple cysts. Conservative operative procedures have been advocated for many years 4,5,7,12. On the other hand, as liver surgery has improved ‘radical’ methods have been performed with good results 4,6. Judging from our experience, satisfactory results can be achieved with both tactics. The postoperative morbidity was the same in groups A and B and complications were easily treated. Although the hospital stay was longer in group A, there were more complicated cysts in this group 3,6,9,12. Compared with reported mortality rates around 3%, our 1.36% rate was very low 6. The patients in group A who died had multiple cysts and pulmonary insufficiency. Our low morbidity and mortality rates were probably due not only to early diagnosis and proper technique but also to good perioperative intensive care. Lastly, the 8.2% reccurence rate in our conservative group is in line with previous reports 6,7. In this regard the perioperative administration of anthelminthic therapy may be beneficial 5. We see little place for unconventional treatments like percutaneous inactivation or laparoscopic surgery in managing multiple hydatid cysts 13,14.
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