Management of an Esophago-pleural Fistula after Emergent Endoscopic Variceal Injectional Sclerotherapy: A Case Report And Literature Review

Abstract

A 48-year-old man developed sudden-onset haematemesis and melena after decompensated posthepatitic cirrhosis. Endoscopic variceal injectional sclerotherapy was emergently performed. However, the patient developed esophago-pleural fistula, empyema, and liver failure. He thus received symptomatic treatments and nasojejunal feedings, which failed to restore the nutrition as the gastroesophageal reflux exacerbated the hydrothorax. Percutaneous endoscopic gastro-jejunal (PEG-J) was therefore carefully performed for enteral nutrition support. The patient had recovered from the fistula at a six-month follow-up, which allowed the resumption of an oral diet. Our literature review revealed that PEG-J is a feasible approach to treating esophago-pleural fistula, a rare but lethal complication of endoscopic sclerotherapy.

Introduction

Acute variceal bleeding is the most lethal complication of portal hypertension (1). Over the past several decades, endoscopic variceal injectional sclerotherapy (EVIS) has gained wide popularity for the initial control of active esophageal variceal bleeding (EVB) (2). The technique of EVIS is achieved by injection of sclerosants to cause venous obturation and haemostasis, which has the advantages of a lower mortality and reduced incidence of hepatic encephalopathy compared with conventional portosystemic shunts (3).

However, due to anatomical factors and its reliance on the endoscopist's experience, EVIS carries a risk of complications, such as pleural effusions and mediastinitis. Among those, esophago-pleural fistula (EPF) is a rare but fatal complication of EVIS with undeniably poor outcomes (4). To date, there have only been four studies, including this one, that have reported the management of EPF caused by EVIS.

This article emphasizes the role of percutaneous endoscopic gastro-jejunal (PEG-J) in the treatment route for EPF and further includes a literature review of cases to support a comprehensive understanding of the treatment experience.

Case Report

A 48-year-old man presented to the emergency room with severe sudden-onset haematemesis and melena. He had a three-year history of decompensated hepatitis B cirrhosis and repeated episodes of upper gastrointestinal bleeding. The patient was classified as Child-Pugh B with an estimated blood loss of 1.5 L. He immediately underwent endoscopic variceal injectional sclerotherapy performed by experienced endoscopists (Fig. 1). Four varicose veins were found on the fundus, and tissue adhesive was injected. Five varicose veins, including one ruptured, were found on the lower esophagus with a red-color sign, and lauromacrogol injection was applied.

Figure 1.

Emergency endoscopic sclerotherapy for esophageal varices.

The endoscopic surgery was completed smoothly, followed by the administration of acid inhibition, anti-digestion, blood transfusion, antibiotics, and rehydration therapy. However, on postoperative day 6, the patient suffered from sudden dyspnoea and acute retrosternal pain, and bedside chest X-ray showed left hydropneumothorax and pneumonia (Fig. 2).

Figure 2.

Bedside chest radiograph of the postoperative patient.

Thoracentesis was performed, and a subsequent microbial culture revealed Klebsiella pneumonia. The respiratory symptoms were alleviated after hydrothorax drainage, but the infection persisted [maximum temperature (T_max): 39.0 °C, white blood cell (WBC): 17.6×10⁹/L, C-reactive protein (CRP): 97.5 mg/L], accompanied by aggravated liver function [total bilirubin (T-Bil): 296.5 μmol/L, direct bilirubin (D-bil): 189.8 μmol/L, albumin (ALB): 23.2 g/L] and a Nutritional Risk Screening (NRS 2002) score of 1 point and body mass index (BMI) of 30.0 kg/m². Chest computed tomography (CT) demonstrated encapsulated effusion in the left pleural cavity and mediastinum, along with bilateral pneumonia (Fig. 3). These findings suggested the progression to esophago-pleural fistula, empyema, and acute-on-chronic liver failure.

Figure 3.

Postoperative chest CT image.

Sufficient imipenem, liver protectants, proton pump inhibitors, and parenteral nutrients were further administered daily. After eight days of medication, the patient was transferred to a hospital in Shanghai at his request and then sent to a local hospital for supportive treatment shortly after the onset of gastrointestinal haemorrhaging.

Three months later, the patient was readmitted to our ward. On an examination, his nutritional status was quite poor (BMI: 22.4 kg/m², ALB: 28.2 g/L, NRS 2002 score: 4 points), although the liver function had markedly improved (Child-Pugh class A). After confirming the partial recovery of the esophageal fistula by esophageal angiography, a nasojejunal tube was placed to establish an enteral nutrition pathway. However, the hydrothorax became aggravated during jejunal nutrition, possibly due to the gastroesophageal reflux. To address this, a nasogastric tube was further placed endoscopically for decompression (Fig. 4).

Figure 4.

Endoscopic nasogastric tube placement. Multiple fistulas were seen in the middle and lower esophagus, and varicose veins were seen in the lower segment.

After two months of comprehensive treatments, both the nutritional status and hydrothorax gradually improved (BMI: 23.2 kg/m², ALB: 30.4 g/L, NRS 2002 score: 3 points). Taking the patient's discharge intention and the attendant risk of variceal rebleeding into account, endoscopic placement of PEG-J were smoothly performed to substitute the transesophageal tubes (Fig. 5). Pre-discharge CT showed the markedly absorbed hydrothorax, at which point the drainage tube was removed (Fig. 6).

Figure 5.

Endoscopic gastrostomy combined with jejunal feeding tube placement.

Figure 6.

Pre-discharge chest CT showed that the patient’s left pleural effusion was significantly absorbed compared to the previous one, and bilateral pneumonias were also alleviated.

Six-month follow-up was continued, during which time comprehensive treatment was resumed, including nutritional support, liver protection, antiviral and anti-infection therapy, acid suppression, and gastroprotective drugs. For the enteral nutrition formulations, the patient sequentially received Revilife^Ⓡ for one month, Peptisorb Liquid^Ⓡ for one month, and Ensure^Ⓡ for four months. In addition, a daily late-evening snack containing 200 kcal of carbohydrates was provided. The radiological and endoscopic examinations revealed the closure of the fistulous tract and a good condition of the PEG catheter. The BMI reached 25 kg/m², the serum ALB level increased to 38 g/L, and the NRS 2002 score was 1 point. Afterwards, the catheter was successfully removed, and the patient resumed an oral regimen comprising 600 kcal/day of Ensure^Ⓡ, along with esomeprazole and entecavir tablets, as well as a conventional diet aligned with the principles of hepatocirrhosis.

Regrettably, three months later, the patient died of septic shock and multiple organ failure due to an accidental unhygienic diet.

Discussion

We herein report a male patient who experienced ruptured haemorrhaging from esophagogastric varices due to liver cirrhosis. Subsequent to emergent sclerotherapy treatment, the patient developed a life-threatening complication known as an EPF, which poses considerable challenges in terms of conventional treatment. In light of this situation, we endeavoured to employ PEG-J as a means to facilitate gastric decompression, enteral nutrition, and oral medication administration.

Remarkably, the patient's nutritional status improved with the successful closure of the EPF following the interventions, as evidenced by the improved nutritional indices and the radiological and endoscopic examinations. Therefore, we underscore the potential efficacy of PEG-J as a viable approach for promoting fistula healing and providing enteral nutrition in cases of EPF following EVIS. This approach has potential utility in similar clinical scenarios, including postoperative cases of esophageal or cardia cancer, as well as instances where EPF arises from other causes.

EVB is one of the most feared complications of decompensated cirrhosis and requires urgent and effective treatment (5). EVIS has the advantages of direct vision, reliable haemostasis, and minimal invasion and is considered the first-line therapy for EVB (3). After entering the veins, the sclerosant destroys endothelial cells and causes inflammation, triggering thrombosis in the varicose veins and local mucosa. Within weeks or months, venous thrombosis undergoes granulation and organization, thereby leading to fibrosis of the esophageal wall and diminishment of the varicose veins (6).

Although its usage has been largely replaced by endoscopic variceal ligation due to the relatively higher risk of complications and a similar efficacy profile, EVIS remains a viable therapeutic option for certain patients with EVB, particularly at sites proficient in this treatment modality, such as our institution. The common postoperative complications of EVIS include a fever, post-sternal pain, and pleural effusion, while severe complications, such as esophageal perforation, stenosis, and ectopic embolism, are extremely rare. EPF is a particularly rare complication that can lead to fatal outcomes, including empyema, sepsis, and even multiple organ failure (4). Postoperative EPF of EVIS was first reported by Carr-Locke et al. in 1982, whereas postoperative empyema was originally described by Chertow et al. in 1991 (7,8). Despite having a very low prevalence of 0.5-5% and often occurring 7-14 days after surgery, EPF has an extremely poor prognosis (9). In most cases, EPF was caused by ulceration from sclerosant extravasation and subsequent chemical necrosis of the esophagus wall, rather than direct mechanical injury. This complication has been linked to conditions such as a high dose of sclerosants, extravascular injection, emergency surgery, and severe liver disease.

A PubMed search using the MeSH terms (‘endoscopic injection sclerotherapy' or ‘endoscopic variceal injectional sclerotherapy') and ‘esophago-pleural fistula' revealed three case reports, one of which was unfortunately un-downloadable. In one case, the authors described a 4.5-year-old boy with extrahepatic portal vein obstruction and the receipt of EVIS within 2 weeks. An examination revealed unilateral pleural effusion and empyema cavity. Initial chest tube drainage, antibiotics, and oral feeding were not very effective, as he developed a right-sided EPF. Later, a nasojejunal tube was used, and the fistula was gradually ameliorated (10). In another case, an 81-year-old man with schistosomiasis-induced hepatic cirrhosis was hospitalized with upper gastrointestinal haemorrhaging. EVIS was conducted, consequently inducing an EPF. The patient was then carefully treated with endoscopic self-expandable metallic stent placement, nasojejunal nutrition, and other comprehensive procedures. After six-month follow-up, he had been entirely cured with no recurrence (11).

These patients, including the case in our report, shared similar features. Conservative therapy is extensively employed for EPF, due to the high prevalence of surgical failure linked with embrittlement and oedema of the postoperative tissue. Treatment in the acute phase focuses on closed chest drainage, anti-infection and nutritional support. Enteral nutrition has merits over parenteral nutrition in infection control and fistula healing and thus is crucial in patients with progressive liver disease. To this end, a nasojejunal tube can be applied for enteral feeding of EPF and can be paired with esophageal stents (12). However, due to the transoesophageal feature and the proximity to the varicose veins, nasojejunal tubes carry a re-bleeding risk and cause discomfort to patients. In contrast, PEG-J has the benefits of avoiding varices with a low risk of re-bleeding, minimal mobility, low incidence of reflux, and patient acceptability (13).

This case offers invaluable insights into the careful management of EPF following EVIS. First, meticulous monitoring of postoperative patients is essential. The presence of a fever, chest discomfort, escalating pleural effusion, or worsened hepatic dysfunction may indicate transesophageal necrosis and perforation. Second, prioritizing enteral nutrition while preventing associated complications, such as reflux and catheter-related infections, is crucial. The specific circumstances dictate whether PEG-J or nasojejunal tube placement is preferred. Third, given the potential complications arising from decompensated cirrhosis, supportive therapy should be intensified. Finally, a compromised hepatic reserve function portends an unfavorable prognosis, as exemplified by the present patient who, despite recovering from EPF, succumbed to hepatic failure after an intestinal infection.

Conclusion

Our findings suggest that PEG-J may be a feasible option for promoting fistula healing and enteral nutrition for EPF patients after EVIS.

Ethics approval for the present study was approved by the Human Research Ethics Committee of our hospital (2023-030RS-01), and written informed consent was obtained from the patient's family.

The authors state that they have no Conflict of Interest (COI).

Financial Support

This work was supported by the Medical Health Science and Technology Project of the Zhejiang Provincial Health Commission (No. 2018KY681) and the National Science Foundation of Ningbo City (No. 2022J205).