With its various genotypes, Echinococcus granulosus is the etiologic agent of cystic echinococcosis (CE) of the liver, a disease that is usually asymptomatic, and whose diagnosis is usually made incidentally. As the disease evolves, its complications include the development of communications between the cyst and biliary tree, which can lead to cholangiohydatidosis,1 rupture into the peritoneal cavity, and the subsequent seeding of hydatid material.2,3 Furthermore, the progressive complications may present in combination with others, such a cyst communicating with the biliary tract that ruptures into the peritoneal cavity, which will lead to acute biliary peritonitis (ABP) (Fig. 1).
It is estimated that the rupture of CE into the peritoneal cavity is associated with superficial cyst locations and diameters greater than 10 cm, which may occur in 1%–16% of CE of the liver.4
The aim of this study was to determine the clinical characteristics and postoperative complications (PC) of patients who had undergone surgery for ABP secondary to hepatic CE that had ruptured into the peritoneum.
We conducted a case series and follow-up of consecutive patients operated on at the Regional Hospital of Temuco, Chile due to CE of the liver complicated by ABP (January 2008 to March 2023), with no exclusions. All patients had a general lab work-up, chest x-rays, ultrasound and computed tomography.
The outcome variable was PC (dichotomized as yes/no according to the Clavien-Dindo proposal) registered within 3 months after surgery. Other variables of interest were: mortality and recurrence; comorbidities (Charlson index5); cyst diameter and location; and type of surgery performed.
All patients were treated by laparotomy under general anesthesia and were administered antibiotic therapy with ceftriaxone and metronidazole.
After laparotomy, the peritoneal fluid was drained, hydatid material was removed, and lavage was performed with saline solution. Subsequently, the cystic lesion(s) were treated with either total or partial pericystectomy, or liver resection. The communications between the duct and biliary tree were sutured, and the residual cavity was resolved with omentoplasty. Lastly, a drain was placed in the right hypochondrium. The surgical specimens were sent for pathological study. The minimum follow-up was 18 months, with general laboratory studies and imaging tests. Data masking was implemented, and descriptive statistics were applied.
During the study period, 13 patients were treated with an average age of 43.5 ± 19.1 years; 61.5% were male, and 69.2% had comorbidities. One patient had a history of surgery for CE of the liver, and 38.5% had a Charlson Comorbidity Index (CCI) ≥ 3.
The laboratory test results demonstrated values within normal limits (with the exception of alkaline phosphatases), except for the maximum levels of total leukocyte count, total bilirubin, and both transaminases, as well as glycemia and creatinine in patients with comorbidities. The lesions were WHO stage CE4, and the most frequent location was the right lobe (46.2%), although echinococcosis was also found in other locations in 53.8% of our patients. The most frequently used technique was pericystectomy (Table 1).
Distribution of clinical variables of the patients in the study (n = 13).
| Variable | N cases | % |
|---|---|---|
| Comorbiditiesa | ||
| None | 7 | 53.8 |
| Liver hydatid cyst | 4 | 30.8 |
| HTN | 3 | 23.1 |
| T2DM | 2 | 15.4 |
| Liver cirrhosis | 1 | 7.7 |
| CCI (points) | ||
| 0 | 7 | 53.8 |
| 1–2 | 1 | 7.7 |
| 3–4 | 4 | 30.8 |
| ≥5 | 1 | 7.7 |
| Other locations | ||
| Liver (and ruptured cyst) | 4 | 30.8 |
| Peritoneum | 3 | 23.1 |
| Ruptured cyst location | ||
| Right lobe | 6 | 46.2 |
| Center liver | 4 | 30.8 |
| Left lobe | 3 | 23.1 |
| Observed biliary communications | ||
| 1 | 6 | 46.2 |
| ≥2 | 7 | 53.8 |
| Type of surgery | ||
| Partial pericystectomy | 4 | 30.8 |
| Total pericystectomy | 6 | 46.2 |
| Bisegmentectomy II-III | 3 | 23.1 |
| Other simultaneous surgeriesb | ||
| None | 3 | 23.1 |
| Cholecystectomy | 6 | 46.2 |
| Resection of peritoneal cysts | 3 | 23.1 |
| Choledocostomy | 2 | 15.4 |
| Cholecystectomy + choledocostomy | 1 | 7.7 |
HTN: hypertension.
T2DM: type 2 diabetes mellitus.
The mean main cyst diameter, surgical time and hospital stay were: 18.3 ± 5.9 cm; 116 ± 32 min and 6.7 ± 3.1 days, respectively.
The postoperative complication rate was 38.5%, including 3 cases of surgical wound infection (23.1%), one atelectasis, and one cardiac arrhythmia, all of which were Clavien-Dindo I or II (I: 23.1%; II: 15.4%). No reoperations were needed, and mortality was 0%. In the 3 cases of concomitant peritoneal echinococcosis, albendazole (10 mg/kg) was prescribed for 3 months. With an average follow-up period of 77 ± 33 months, no recurrence was observed.
Between 7% and 38% of ruptured hepatic CE have been associated with a history of blunt abdominal trauma,6,7 a fact that did not occur in our series, although it has been reported.8 There was also no anaphylaxis, which has been reported in up to 25% of cases.7 This low frequency can be explained by the fact that these cysts present with communications to the biliary tract, with potentially impaired viability due to the lytic effect of bile as a natural detergent.9 Concomitance with another abdominal CE may reach 30%,10 which in our experience was 23.1%.
Regarding the study variables, the previously published evidence provides the following results: 5–18 days of hospitalization;6–8 35%–43% PC;6,10 and up to 24% mortality and intra-abdominal recurrence. Median follow-up times are 63–78 months.6,8,10 Although some series have reported no recurrence, their follow-ups are <36 months.7 These figures are somewhat worse than our experience (5 days; 38.5%; 0% and 0%, respectively, with a median follow-up of 77 months).
In short, and to conclude, the rupture of CE towards the peritoneal cavity, while rare, poses a diagnostic-therapeutic challenge and should therefore be included in the differential diagnosis of acute abdomen in endemic areas of echinococcosis. Timely diagnosis and appropriate urgent surgery can reduce postoperative morbidity and mortality, which are still higher than in cases of elective surgery for uncomplicated CE of the liver. This is a single-center, regional case series with a considerable number of cases compared to the very limited existing evidence of this complication of evolved disease. In terms of postoperative complications, mortality and recurrence (with long-term follow-ups), our results are better than the results reported by the few series with similar samples and follow-up periods.
FundingPartially funded by the DIUFRO DI23-0020 and DIUFRO DI23-0073 projects, Dirección de Investigación y Desarrollo de la Universidad de La Frontera.
