Ann Thorac Surg 2001;72:1883-1886
© 2001 The Society of Thoracic Surgeons
Original article: general thoracic
Thoracic rupture of hepatic hydatidosis (123 cases)
El Hassane Kabiri, MD*a,
Abderahman El Maslout, MDa,
Abdellatif Benosman, MDa
a Department of Thoracic Surgery, Ibn Sina University Hospital, Rabat, Morocco
Accepted for publication August 6, 2001.
* Address reprint requests to Dr Kabiri, 12 C, 15 Rue Alloysia, Riad 10100, Rabat Maroc, Morocco
e-mail: hassankabiri{at}yahoo.com
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Abstract
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Background. Hydatid disease is frequently endemic in countries with poor environmental sanitation and in geographic areas where interaction between humans and animals is common. Pulmonary complications result from the proximity of hydatid cysts in the liver and the diaphragm.
Methods. The medical records of 123 patients, with established hydatid disease manifesting abnormal chest roentgenograms, were retrospectively analyzed for the period January 1990 to December 1999.
Results. Chest roentgenogram and abdominal ultrasound provided a correct preoperative diagnosis in 108 patients (87.8%). Expectoration of bile, demonstration of fistula by ultrasound, expectoration of cyst contents, and additional ultrasound or imaging findings were the criteria used to establish the preoperative diagnosis. The remaining 15 cases were confirmed at operation. Men outnumbered women nearly 3:1. Mean age was 36.2 years. Pulmonary resection was performed in 67 cases. Sixty-eight patients presented with a bronchobiliary fistula (55.3%). Morbidity rate was 14.6% and mortality rate was 8.9%.
Conclusions. Thoracotomy offers adequate simultaneous access to both the chest and hepatic lesions with acceptable morbidity and mortality. Endoscopic sphincterotomy undertaken preoperatively is useful in reducing biliary complications.
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Introduction
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Hydatid disease is an infection caused by ingestion of eggs from the dog tapeworm Echinococcus granulosis [1]. It can also occur in cattle-raising and sheep-raising areas of the world. The parasite lives in the intestinal tract of carnivores. Its head is composed of a double-crown of hooklike structures, and three or four rings form the body. The eggs reside in the last ring and are eliminated with the feces. The eggs, which contaminate grazing fields and irrigated land, are consumed by herbivores. Humans become affected from water, food, or direct contact with carnivores. Once the eggs reach the human stomach the hexacanth embryos are released. These larvae undergo encystation in the small intestine, penetrate the intestinal wall, and are carried by mesenteric vasculature to various sites within the body including the liver, lung, and, rarely, the kidney, the brain, or other sites. A single fluid-filled cyst forms and contains multiple protoscoleces, one of which is capable of creating a new cyst. Symptoms develop when the cyst enlarges sufficiently to be a space-occupying mass. The liver is the most common location, resulting in abdominal pain and signs of cholestasis.
Rupture of a hydatid cyst may occur spontaneously or after blunt trauma and can precipitate anaphylactic shock. The spillage of protoscoleces leads to multiple daughter cysts, which develop months to years later. Intrathoracic rupture is a rare complication with an incidence of 0.6% to 16% of cases [2–4]. Bronchobiliary fistula is a serious complication and requires prompt surgical treatment.
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Patients and methods
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During a 10-year period (January 1990 to December 1999), 123 patients with hydatid cyst of the liver that ruptured into the thoracic cavity underwent surgical treatment at Ibn Sina University Hospital, Rabat, Morocco. Twenty-five patients (20.3%) had previous surgical treatment of hepatic hydatid cyst 6 to 14 years before developing a pulmonary complication. There were 91 men (74%) and 32 women (26%). The mean age at the time of surgery was 36.2 years old (range 12 to 71 years). The pulmonary extension occurred on the right side in 120 patients and was left sided in 3.
Clinical presentation was essentially pulmonary, dominated by expectoration of bile in 50 patients (40.6%), thus confirming the presence of a bronchobiliary fistula. Abdominal signs were rarely encountered. Two patients presented with cutaneous fistula, draining pus and bile (Table 1).
Chest roentgenogram was performed in all patients with identification of an elevated right diaphragm in 27 cases, air-fluid levels in 18, basal infiltrations in 32, and pleural effusions in 56. Abdominal ultrasound was abnormal in all but 3 patients. Findings were 120 hydatid cyst ranging in size between 2.5 and 18 cm. A fistula was identified in 20 patients and common bile duct stones were seen in 6 patients.
Immunoelectrophoresis and indirect hemagglutination was positive in 55 of 74 patients tested (74.3%). Laboratory examination revealed 15 patients with hemoglobin levels less than 8 g/100 mL, leukocytosis greater than 14,500 mL3 in 25 patients, hyponatremia in 9, and hypoproteinemia in 5.
Bronchoscopy was undertaken in 54 patients and demonstrated the exit of bile or vesicles. Only 25 patients underwent computed tomography, which demonstrated bronchectasis in 16, abscess in 7, and atelectasis in 2.
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Results
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Surgical results
All patients underwent posterolateral thoracotomy in the sixth intercostal space (120 right, 3 left). In 8 patients a secondary eighth intercostal space thoracotomy, using the same skin incision, was performed to facilitate freeing of the lung from the diaphragm.
The surgical steps proceeded as follows: evacuation of the pleural cavity, pulmonary decortication, localization of the diaphragm breach, dissection of the diaphragm and liver, treatment of the hepatic lesions, closing of the diaphragm, treatment of the pulmonary pathology, and drainage of both the chest and subphrenic space. Six patients had simultaneous laparotomies to remove common duct stones [2], repair an intraoperative injury to the inferior vena cava [1], and resect an enlarged gallbladder [3]. Subhepatic drains were placed in all 6 patients. The breach in the diaphragm was identified in all cases, enlarged for access to the liver if necessary, and a subphrenic drain placed before closure of the diaphragm. We rarely used scoliocidal products. Postoperative pleural drainage was maintained for 2 to 16 days (mean 5.7 days) and abdominal drains were kept in place for a minimum of 7 days or removed when the output of bile or fluid was less than 5 mL (range 7 to 45 days; Table 2).
Series results
Clinical findings, chest roentgenogram, and abdominal ultrasound permitted a correct preoperative diagnosis of hepatic hydatid cyst and intrathoracic rupture in 108 patients. The remaining 15 patients were diagnosed intraoperatively. Criteria used to arrive at a correct preoperative diagnosis were: expectoration of bile (50 cases), ultrasound demonstration of a fistula (20 cases), expectoration of cyst contents and other ultrasound abnormalities (24 cases), and imaging studies in those with hemoptysis (14 cases). Intraoperative complications occurred in 15 cases: hemorrhage exceeding 1,000 mL, but not requiring transfusions (8 cases); important biliary leaks (5 cases); laceration of the inferior vena cava controlled through a laparotomy (1 case); and death from severe hypoxemia and secondary cardiac arrest (1 case). Postoperative complications occurred in 18 patients (14.6%) classified as thoracic (11 cases), biliary (2 cases), and general (5 cases) (see Table 3). Eleven deaths occurred and are summarized in Table 4. The mean hospital stay was 20 ± 5.2 days (range 7 to 105 days). Mean follow-up was 8.5 years (range 1 to 9 years). The patients were seen systematically at the first, third, and 12th month after operation for clinical, serologic, and radiologic evaluations. Late complications or recurrences were noted in only 1 patient who presented with a thoracic cutaneous fistula 2 years after operation. Chest roentgenogram in that patient demonstrated a pleural effusion, which was treated with tube drainage for 33 days with a successful outcome.
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Comment
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Intrathoracic rupture of hepatic hydatid cyst is a rare but severe condition causing a spectrum of lesions to the pleura, lung parenchyma, and bronchi. Cyst erosion is associated with pericystic inflammation. Adhesion formation determines whether the rupture is confined to lung parenchyma or the free pleural space, or both. Bronchobiliary fistula leads to hemoptysis and cyst expectoration. The clinical presentation is predominately pulmonary, with abdominal symptoms being less frequent [3, 4]. Cough, expectoration, and dyspnea are present in 30% of cases. Jaundice implies rupture into a bile duct with obstruction. Chest roentgenogram findings can be typical or not; frequently, a right lower lobe opacity or pleural effusion is seen. Abdominal ultrasound is necessary to show the morphology of the liver and possible biliary tree obstruction or stones. Computed tomography aids in differentiating the relationship among the cyst, blood vessels, and bile ducts, and identifying other possible sites and assisting in the choice of surgical approach [2–5]. Because most of our patients were seen on an emergency basis computed tomography was rarely performed (in only 25 cases). Bronchoscopy, when performed, may show bile, daughter vesicles, or fragments of cyst membrane. It also permits assessment of the severity of inflammatory process. Many factors may promote the intrathoracic occurrence of a hepatic hydatid cyst: the pressure gradient between the pleural and abdominal cavities favors a thoracic direction; compression and ischemia of the diaphragm secondary to inflammation about the cyst; and the chemical action by bile on the diaphragm, lung, and pleura [3, 4].
In 1987 Mesteri and colleagues [6] enhanced Deve’s classification into four types: type I: direct fistulization of the cyst into the bronchi with small, I-A, or large fistula, I-B; type II: intrapulmonary parenchymal collection without fistula, II-A, or with large fistula, II-B; type III: intrapleural encysted collection without, III-A, or with bronchitic fistula, III-B, or parietal fistula, III-C; and type IV: rupture into the pleural cavity with pleural effusion (hydatidothorax), IV-A, or a secondary pleural hydatidosis, IV-B. Hyponatremia and hypoproteinemia are usually related to bile leakage and should be corrected preoperatively. Hypoxemia is frequently noted. Postoperative results are directly related to correction of fluid and electrolyte imbalance, aspiration of the bronchi, respiratory physiotherapy, and antibiotic agents. Other authors use only laparotomy to treat this disease [2, 3, 7]. We believe that laparotomy alone is insufficient. Bile is destructive and bronchiectasis will necessitate later operation. Because both sides of the diaphragm can be adequately addressed in most cases, we do not concur with those who recommend thoracotomy only for pleural or lung parenchymal involvement [2, 8, 9]. Our position is supported by other authors [4, 6, 10, 11]. Some authors have recommended endoscopic sphincterotomy for relief of biliary obstruction [12]. This technique may be a reasonable alternative to laparotomy. Higher complication rates with endoscopic sphincterotomy were reported by Xanthakis and coworkers (39.4%) [13], Sakhri and colleagues (65.9%) [3], Karydakis and colleagues (37.2%) [7], and Kilani and colleagues (31.3%) [4], compared with only 14.6% in the present series. The lower rate in our series perhaps may have been lessened by using a thoracic approach.
The mortality rate of this disease is high, varying from 9% to 43% [3, 4, 7, 9, 14]. In the present series the mortality rate was 8.9%. Except for type IV-B and rarely in type IV-A, medical adjuvant treatment is never required for a ruptured intrathoracic hepatic hydatid cyst. It is indicated when dissemination is confirmed and total resection of the cysts is not possible. Albendazole 400 mg twice daily for 6 months is the current recommended therapy when indicated. In endemic areas, prophylactic measures should always be taken.
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Acknowledgments
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We thank Richard G. Fosburg, MD, for editing this article for language.
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References
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Abstract
Introduction
