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Ann Thorac Surg 2008;86:1690-1693. doi:10.1016/j.athoracsur.2008.03.023
© 2008 The Society of Thoracic Surgeons

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Case Reports

Multidisciplinary Therapy of a Large Esophago-Pericardial Fistula Arising From Barrett's Esophagus

Firas Ali, MDa,*, Ahmed Mostafa, MDa, Antoine Charachon, MDb, Mehdi Karoui, MD, PhDc, Daniel Loisance, MDa, Matthias Kirsch, MD, PhDa

a Department of Thoracic and Cardiovascular Surgery, Henri Mondor Hospital, Créteil, France
b Department of Gastrology, Henri Mondor Hospital, Créteil, France
c Department of General Surgery, Henri Mondor Hospital, Créteil, France

Accepted for publication March 14, 2008.

* Address correspondence to Dr Ali, 51 Avenue du Maréchal de Latter de Tassigny, Crétéil, 94000, France (Email: matthias.kirsch{at}hmn.aphp.fr).


    Abstract
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 Abstract
 Introduction
 Comment
 References
 
Esophago-pericardial fistula is a rare complication of benign esophageal pathologies. We present the case report of a patient with Barrett's esophagus complicated by an esophago-pericardial fistula managed by a multi-therapeutic approach to close the fistula using an autologous pericardial patch; placement of a coated, expandable, metallic esophageal stent; and a feeding jejunostomy.


    Introduction
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 Abstract
 Introduction
 Comment
 References
 
Esophago-pericardial fistula (EPF) is an extremely rare and severe complication of malignant, benign, and traumatic pathologies of the esophagus with a high mortality rate [1, 2].

Here we describe the first case of successful treatment of Barrett's esophago-pericardial fistula using a combination of three different therapies: (1) temporary stenting of the distal esophagus, (2) closing of the fistula using a patch of autologous pericardium through a median sternotomy, and (3) a feeding jejunostomy.

A 46-year-old man, with smoking and alcohol as the main risk factors, presented to the emergency room with a 3-week history of nonradiating retrosternal chest pain. Clinical and biological examination showed diminished heart sounds, a raised temperature (37.8°C), and a white blood cell count of 27,000/mL. A chest roentgenogram (Fig 1A) showed an image of pneumopericardium in which transthoracic echocardiography was hampered by the pericardial trapped air. A 64-row multi-slice computed tomographic scan coupled with water-soluble contrast agent (Fig 1B) detected the presence of communication between the distal esophagus and the pericardial sac. The upper gastrointestinal tract endoscopic view showed a long Barrett's esophagus affecting the last 10 cm of the esophagus, and the existence of a large fistula (3 x 2.5 cm) between the esophagus and the pericardium, which was located approximately 2 cm above the gastroesophageal junction.


Figure 1
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Fig 1. (A) Evidence of pneumopericardium on the chest roentgenogram. (B) Multi-slice computed tomographic scan showing (1) trapped air in the pericardial sac, (2) the fistula, and (3) the thickened wall of the esophagus.

 
Histologic analysis of tissue samples harvested at both the Barrett's esophagus and at the level of the fistula showed a glandular mucosal high-grade dysplasia with no evidence of malignancy.

We decided to operate on the patient in cooperation with the local general surgery and gastroenterology departments to perform the following:

1 First, endoscopic stenting of the distal esophagus was achieved using a flexible and coated self-expandable, metallic stent (Fig 2A). The stent measured 16 cm in length and 2.2 cm in diameter (Hanarostent covered esophageal stent with Shim Anti-reflux valve [M.I. Tech Co Ltd, Seoul, Korea]), which is similar to those used for the management of a malignant esophageal fistula. The stent was long enough to cover all of the length of the Barrett's esophagus, and even lower down to the stomach. The stent was fixed by nine large, metallic clips at its upper and lower parts. This was followed by the placement of a nasogastric tube.
2 Second, a median sternotomy was performed, which was followed by stringent washing of the pericardium. Fibrin deposits covering the heart were removed. The fistula was closed by fixing an autologous pericardial patch (Fig 2B) to the tissues surrounding the ulcer using several separated 5-0 Prolene stitches (Ethicon, Somerville, NJ) on a beating heart, without cardiopulmonary bypass. Pericardial drainage using six Redon tubes (Health Brilliant Medical Instruments Co Ltd, Nanjing, Jiangsu, China) was then installed before closing the sternum.
3 Last, a small laparotomy was performed for a feeding jejunostomy.


Figure 2
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Fig 2. Perioperative views of (A) coated, self-expandable, metallic stent views (1) through the fistula after lifting the heart, (2) of the fistula, and (3) of the inferior vena cava. (B) Patch of pericardium fixed on (1) the fistula of (2) the inferior vena cava.

 
The patient was transferred while intubated to the intensive care unit with 6 µg/kg/min of dobutamine. After 12 hours, artificial ventilation was weaned. The postoperative medical treatment combined proton pump inhibitors (ie, Pantoprazole [Inipomp; Sanofi Synthélabo, Paris, France]) and a large spectrum of antibiotics including Fluconazole (Triflucan; Paris, France) for 3 weeks; Amikacine (Amiklin; Bristol Myers-Squibb, Paris, France) for 4 days, followed by Gentamicine (Gentalline; Shearing Plough, Levalois, France) for 4 more days; Piperacilline (Tazocilline; Wyeth-Lederle, Paris, France) for 3 days, followed by Amoxicilline/clavudanic acid (Augmentin; Smith Kline Becham, Nanterre, France) for 2 weeks.

All drainage tubes were mobilized progressively between postoperative days 14 and 18, as performed in our standard post-sternotomy mediastinitis management [3], except that tube number 4 that was left in place for 21 days because of microbiological evidence of Candida albicans in the drained fluid. C-reactive protein and white blood cell count came to normal values on postoperative day 14.

At night on postoperative day 17, the patient presented with hyperthermia (39.5°C), and blood cultures showed the presence of klebssiella pneumonia and Enterobacter cloace; the evolution was favorable by 2 weeks coverage of antibiotic therapy, which was confirmed by culture and sensitivity tests.

During hospitalization, the weekly follow-up echocardiographic scan showed neither pericardial air nor pericardial collection, and it also confirmed the absence of endocarditis signs. The first follow-up gastrointestinal tract endoscopy was performed on postoperative day 24, which showed the incomplete healing of the fistula, some fibrin deposition on the pericardial patch, and a partial migration of the stent into the stomach, which was still partially covering the fistula. The second follow-up gastrointestinal tract endoscopy was done at 1 month after surgery (Fig 3), which showed complete healing of the fistula; the stent was removed during that same procedure. Normal feeding was then started, and the jejunostomy was closed on postoperative day 40. After 4 months the patient is still alive and he is in good general condition.


Figure 3
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Fig 3. Gastrointestinal tract endoscopic control at 1 month shows total healing of (1) the fistula.

 

    Comment
 Top
 Abstract
 Introduction
 Comment
 References
 
Esophago-pericardial fistula is a classic complication of malignant pathologies of the esophagus [1]. However, only rare cases [2] of fistulas complicating benign affection of the esophagus without prior surgery, endoscopy, or fibrillation ablation procedures have been described in the literature [4].

Barrett's esophagus is a benign esophageal pathology characterized by the presence of metaplastic intestinal mucosa of the lower part of the esophagus, which is in continuity with the gastric mucosa [5, 6]. In addition, there is an inflammatory infiltration of the esophageal wall that is at risk of superficial ulceration or erosion into a mural vessel, such as the aorta or the heart, and possibly perforation into the pleural space or the pericardial sac [5, 7].

Unexpectedly this patient presented with no signs of general sepsis related to his purulent pericarditis, and no specific features compared with other reports [7]. The diagnosis was rapidly achieved by chest roentgenogram, computed tomographic scan and gastrointestinal tract endoscopy.

We adopted a unique therapeutic approach combining closure of the fistula and its temporary exclusion. The direct closure of the fistula was impossible because it was 3 x 2.5 cm large and would certainly have lacerated the inflammatory tissues or would have stenosed the distal esophagus, or both. The use of a Dacron patch (DuPont, Wilmington, DE) [7] was not deemed appropriate in an infected area, and the bovine pericardial patch was not as thick as required. The autologous pericardium [7] seemed to be the most appropriate substitute in such a situation. Exclusion of the fistula was performed by temporary stenting [4] of the esophagus for 1 month. A feeding jejunostomy was left in place for 40 days. This approach probably protected and favored the complete healing of the fistula [8].

Because esophageal biopsy specimens showed glandular mucosal high-grade dysplasia, the need for further esophagectomy will be considered for this patient.


    References
 Top
 Abstract
 Introduction
 Comment
 References
 

  1. Luthi F, Groebli Y, Newton A, Kaeser P. Cardiac and pericardial fistulae associated with esophageal or gastric neoplasms: a literature review Int Surg 2003;88:188-193.[Medline]
  2. Solorzano CC, Livingstone AS. Esophagopericardial fistula in a partially excluded esophagus J Am Coll Surg 2007;198:156-157.
  3. Kirsch M, Mekontso-Dessap A, Houel R, Hillion M-L, Loisance DY. Closed drainage using redon catheters for poststernotomy mediastinitis: results and risk factors for adverse outcome Ann Thorac Surg 2001;71:1580-1586.[Abstract/Free Full Text]
  4. Bunch TJ, Nelson J, Foley T. Temporary esophageal stenting allows healing of esophageal perforation following atrial fibrillation ablation procedures J Cardiovas Electrophysiol 2006;17:435-439.[Medline]
  5. Byrad RW. Barrett esophagus and unexpected death Am J Forensic Med Pathol 2007;28:147-149.[Medline]
  6. Shah S, Saum K, Greenwald BD, Krasna MJ, Sonett JR. Esophagopericardial fistula arising from Barrett's esophagus Am J Gastroenterol 1998;93:465-467.[Medline]
  7. Muller AMS, Betz MJ, Kromeier J, et al. Acute pneumopericardium due to intestino-pericardial fistula Circulation 2006;114:7-9.
  8. Salo JA, Heikkila L, Nemlander A, Lindahl H, Louhimo I, Mattila S. Barrett's esophagus and perforation of gastric tube ulceration into the pericardium: a late complication after reconstruction of esophageal atresia Ann Chir Gynaecol 1995;84:92-94.[Medline]



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