Pleural migration of biliary stent 10 years following treatment of hepatic hydatic echinococcosis: A case report

Abstract

Introduction

Echinococcosis, caused by larval stages of taeniid cestodes, primarily affects the liver and is commonly treated surgically. However, a complication post-treatment is biliary fistula, necessitating interventions like biliary stents. While stent complications are recognized, proximal migration leading to pneumonia is exceptionally rare. This case report details an unusual occurrence of biliary stent migration years after hepatic hydatid echinococcosis treatment.

Case presentation

A 42-year-old patient underwent 2014 surgery for a large hydatid cyst, resulting in a biliary fistula. Endoscopic sphincterotomy and biliary stent placement led to a successful outcome. Lost to follow-up, the patient reappeared in 2022 with basithoracic pain, fever, and a thoracic CT scan revealing transdiaphragmatic stent migration causing basal pneumonitis. Antibiotic therapy and endoscopic stent removal ensued with an uncomplicated recovery.

Clinical discussion

This report emphasizes a rare complication that is proximal migration of a biliary stent 10 years post-initial placement for biliary fistula management. Despite the absence of typical risk factors. We managed a successful endoscopic retrieval. This highlights the importance of vigilance and follow-up for potential complications associated with biliary stent. Unusual presentations, like pneumonitis, underscore the need for awareness and a cautious approach.

Conclusion

The primary complication following surgical intervention for hepatic hydatid cysts is the development of an external biliary fistula, necessitating the use of biliary stents for treatment. Given the rarity of complications observed in our case, the removal of stents post-treatment for biliary fistula becomes crucial, underscoring the significance of vigilant follow-up care.

Highlights

• •Biliary stent are a widely used treatment for persistent biliary fistula.
• •Proximal migration is a rare complication of this procedure.
• •It usually occurs in patient with cholangiocarcinoma rarely for non-malignant disease such as hydatic echinococcosis.
• •Proximal migration causing a pneumonia is an extremely unusual complication.
• •Endoscopic management associated or no with surgery is the main treatment.

1. Introduction

Echinococcosis or hydatid disease (HD) is a zoonosis caused by the larval stages of taeniid cestodes belonging to the genus Echinococcosis [4]. The liver is the main site of HD involvement. The treatment of hepatic HD is mostly surgical; one of the complication that can occur following the treatment of the hydatid cyst is biliary fistula [1,2].

Biliary stents are indicated in the treatment of biliopancreatic junction pathology such as biliary fistula. Typically reported complications include pancreatitis, gastrointestinal bleeding, prosthesis perforation, prosthesis stenosis, and cholecystitis due to reflux, regarding prosthetic migration it is accounting for 5 % of complications [3]. Proximal migration complicated by pneumonia is even rarer.

We report this unusual case about a rarely described complication following treatment of hepatic hydatic echinococcosis years after the management of biliary fistula with biliary stent.

This case report has been reported in line with the SCARE Criteria [5].

2. Case report

A 42-year-old patient, with no history of cardiopulmonary or other disease, who underwent surgery in 2014 for a large hydatid cyst of the dome of 10*12 cm size localized in 5 and 8 hepatic segment, presented with a connection with a collateral of right hepatic duct and a proximity with the right hepatic artery, the patient had a resection of the protruding dome. Postoperative course was complicated by a biliary fistula, for which the patient underwent endoscopic sphincterotomy (ES) combined with the placement of a biliary stent, leading to a favorable outcome.

The patient was lost to follow-up thereafter but reappeared in 2022 with basithoracic pain accompanied by fever up to 38.5 °C, without jaundice.

Biological findings showed white blood cells count of (WBC) 14,200/mm³ C reactive protein was 17 mg/L .the patient underwent a X ray chest (Fig. 1) and a thoracic CT scan (Fig. 2), revealing a Trans diaphragmatic migration of the biliary stent causing a basal pneumonitis. The patient received antibiotic therapy, and the stent was successfully removed endoscopically with uncomplicated postoperative recovery.

Fig. 1.

X-ray chest revealing migration of biliary stent.

Fig. 2.

Thoracoabdominal CT scan that shows migration of biliary stent and pneumonia.

3. Discussion

The most common postoperative complication of hepatic hydatid cyst surgery is external biliary fistula. Persistent biliary fistula is not uncommon [3].

The natural Cystic evolution against liver parenchyma erodes the adjacent bile duct and creates communication between the cystic cavity and biliary tree responsible for biliary fistula [1,4].

In cases where there is a connection between the biliary tract and the residual cyst cavity, bile may flow from the biliary tree into the cystic cavity as a result of the pressure gradient formed after surgery [3,4].

A persistent biliary fistula can also be explained by stenosis of the sphincter of Oddi or occlusion of the common biliary duct (CBD) with the hydatid material.

During surgery, we should diagnose this communication either simply when cystic fluid is found to be stained with bile after primary aspiration or when bile flow into the cystic cavity after evacuation of the cyst unveiled by the change in pressure gradient in favor of biliary system.

Because overlooking such a connection during operation happens, it is recommended that a drain be placed to ensure that if a biliary fistula develops, outflow will be controlled [3].

Our patient had a large hydatid cyst 10*12 cm size localized in 5 and 8 hepatic segment. It presented with a connection with a collateral of right hepatic duct (Fig. 3) and a proximity with the right hepatic artery (Fig. 4). The procedure was a roofing of the cyst.

Fig. 3.

Communication with right hepatic duct.

Fig. 4.

Proximity with right hepatic artery.

In the process we diagnosed a biliary fistula that was repaired following a cholecystectomy and a per operative cholangiography. Two drains were placed in the hydatic cavity.

Post-operative follow-up, persistent external biliary fistula was diagnosed and the decision was to treat it with a biliary stent to change the pressure gradient.

The insertion of plastic stent in the biliary duct (BD) is accepted treatment for patients with post-operative biliary fistula. With long-term stent therapy, migration is one of the most important complications, along with stent obstruction and ascending cholangitis [6].

To our knowledge this case is one of few published report of proximal stent migration presenting with pneumonitis [7,8]. This unusual occurrence raises the question of whether there were specific circumstances that facilitated proximal stent migration in this case.

The risk of proximal stent migration has been reported to be increased with malignant strictures, as well as with the use of larger diameter (>10 F) and shorter stents (7 cm) [6].

The stent used in the present case was a 15 cm long stent with a diameter of 7F and therefore none of the reported risk factor was present in our case.

Few studies have focused on the correlation between endoscopic sphincterotomy (ES) before biliary stent and risk of migration. Margulies et al. [9] in a retrospective study reported a higher frequency of stent migration in patients who had stents placed without ES. But in groups study of this report we found out that proximal stent migration was not impacted by endoscopic sphincterotomy.

In a study that aimed to assess the impact of ES prior to biliary stent placement on the risk of post- endoscopic retrograde cholangiopancreatography complications [10].

A comprehensive search of relevant databases and conference proceedings was conducted for studies published between January 1980 and November 2013. Ten studies, comprising 5 randomized controlled trials (RCTs) and 5 observational studies, involving a total of 1623 patients, met the inclusion criteria. The meta-analysis concluded that the placement of a biliary stent without endoscopic sphincterotomy does not appear to be associated with an increased risk of migration.

In a recent retrospective study Emara et al. [11]observed that the migration of stents, both proximal and distal, had an incidence of 8.4 %. The factors independently associated with stent migration included a broad sphincterotomy, a dilated biliary common duct, and biliary balloon dilation. Additionally, stents that remained in place for over a month exhibited a higher susceptibility to migration. Cholangitis and stent obstruction emerged as the most frequently documented complications associated with migrated stents.

According to available data, the performance of ES before stent placement cannot be regarded as a proven risk factor for proximal stent migration.

The unusual degree of migration in our case cannot be explained by known risk factors for stent migration. However we have our hypothesis that is due to hepatic remodelation after hydatic surgery and thus the large amount of time that the patient had a biliary stent on without removal. All this unusual occurrences had converged in a rarely described complication that is our case.

Regarding treatment of this complication, the majority of proximally migrated stents can be retrieved endoscopically; success rates in the range of 85 % have been reported [9,10].

Endoscopic devices frequently used for this purpose are the Dormia basket, extraction balloon, grasping forceps, and the Soehendra stent retrieval device [5,10,11].

In our case the stent was extracted by a stent retrieval devise there was no incident afterwards the post-operative chest X ray (Fig. 5) showed no sign of pneumothorax and the patient had a prophylactic antibiotherapy.

Fig. 5.

X-ray chest after the removal of biliary stent.

The patient was asymptomatic to follow up and declared was satisfied with the outcome.

This case demonstrates that proximal migration of biliary stents does not always present with signs of biliary obstruction and that even pulmonary symptoms are possible. Because the use of stents continues to increase, it is necessary to be cognizant of all potential complications of this form of therapy. To criticize our treatment we should have more restrictive follow up regarding this kind of pathology thus to minimize the risk of complications.

4. Conclusion

The most common postoperative complication of hepatic hydatid cyst surgery is external biliary fistula. Biliary stents are indicated in the treatment of this pathology. They must be performed by a trained gastroenterologist. Our case highlighted an exceptional complication therefore stent removing after the treatment of biliary fistula should be mandatory thus the importance of follow up.

Ethical approval

Our institutions “la Rabta Hospital” and “School of Medicine of Tunis” require no ethical approval for case reports. It is required Author Form for studies on human participants. This is just a case report with written patient approval.

Funding

None.

Author contribution

Souhaib Atri: conceptualization, data curation, redaction, project manager

Elaifia Rany: conceptualization, data curation, redaction, project manager

Amine Sebai: conceptualization, redaction

Hammami.M: resources, visualization

Haddad.A: resources, visualization

Montassar Kacem: supervision, validation, visualization

Guarantor

Elaifia Rany

Research registration number

Not applicable.

Consent

Written informed consent was obtained from the patients for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-inChief of this journal.

Conflict of interest statement

All authors declare that they have no conflicts of interest.