Ann Thorac Surg 2007;84:648-650
© 2007 The Society of Thoracic Surgeons
Case Reports
Esophageal-Left Atrial Fistula: Intraoperative Diagnosis and Management
Minh-Tri Jean-Pierre Nguyen, MDa,
Sébastien Trop, MD, PhDa,
Constantine Soulellis, MDb,
Peter Szego, MD, FRCPCb,
Lorenzo E. Ferri, MD, FRCSCa,*
a Division of Thoracic Surgery, McGill University, Montreal, Quebec, Canada
b Division of Gastroenterology, McGill University, Montreal, Quebec, Canada
Accepted for publication February 28, 2007.
* Address correspondence to Dr Ferri, Montreal General Hospital, Room L9-112, 1650 Cedar Ave, Montreal, Quebec, H3G 1A4, Canada (Email: lorenzo.ferri{at}muhc.mcgill.ca).
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Abstract
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Nontraumatic esophageal-atrial fistulas are usually fatal and are diagnosed postmortem. We report a rare case of intraoperative diagnosis and successful surgical management of an esophageal-left atrial fistula in a 70-year-old woman with a history of brachytherapy, radiotherapy, and chemotherapy for esophageal cancer who presented with progressive dysphagia and hematemesis.
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Introduction
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Non-traumatic esophageal-atrial fistulas are rare entities that become invariably fatal if not diagnosed and treated early. We are aware of only 19 cases (two antemortem diagnoses and one nonfatal outcome) [1] that had been reported prior to a relative proliferation of this condition with the advent of radiofrequency ablation for atrial fibrillation [2]. The most common setting was chronic esophagitis or esophageal carcinoma, resulting in an ulcer penetrating the left atrium. The classical presentation is the clinical triad of hematemesis, acute neurologic symptoms from cerebral emboli and chronic dysphagia [1]. We report the case of an esophageal-left atrial fistula that was diagnosed intraoperatively and successfully managed surgically.
A 70-year-old woman was diagnosed 10 months ago with stage IIB poorly-differentiated carcinoma of the distal esophagus. Due to patient preference, she underwent definitive brachytherapy, external-beam radiotherapy, and chemotherapy. She had complete clinical response. However she had a severe radiation stricture develop that required monthly dilations with repeated biopsies that were benign. She presented to the emergency room 15 days after her last esophageal dilation with progressive dysphagia and hematemesis.
The patient stopped bleeding and an upper gastrointestinal double contrast barium study demonstrated a small traction diverticulum in the mid-lower esophagus (Fig 1). An upper endoscopy revealed a stenotic esophageal lesion associated with a deep ulcer corresponding to the sinus seen on the contrast esophagram (Fig 2). A biopsy of the lesion was performed and was followed by immediate massive hemorrhage from the biopsy site. Attempts at local control using epinephrine and saline were not successful. The patient became unresponsive and required endotracheal intubation. A Sengstaken-Blakemore tube (Rüsch, Duluth, GA) was then inserted and hemostasis was achieved. After resuscitation in the intensive care unit, four intercostal arteries were embolized in the interventional radiology suite, although no active source of bleeding was identified. Under endoscopic guidance, the Sengstaken-Blakemore tube (Rüsch) was removed after 4 hours, which was not accompanied by further bleeding.
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Fig 1. Upper gastrointestinal double contrast barium study revealing a small traction diverticulum in the mid-lower esophagus (arrowhead).
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Fig 2. Upper endoscopy demonstrating a stenotic ulcerated lesion at the lower third of the esophagus representing the esophageal-left atrial fistula later confirmed intraoperatively (arrowhead).
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As the source of bleeding was never definitively identified, our service was consulted and an esophagectomy was proposed. After consent was obtained, an elective Ivor-Lewis esophagectomy was performed. During the laparotomy, a previously placed percutaneous gastrostomy was removed. The stomach was otherwise normal and a 4-cm gastric conduit was prepared based on the right gastro-epiploic artery. As there were no enlarged lymph nodes along the celiac tri-forcation, a standard D1 celiac lymphadenectomy was performed. At a right thoracotomy, identification of the anterior esophageal wall was difficult due to an extensively fibrotic posterior mediastinum. Planes between the pericardium and esophagus were completely obliterated. Therefore, an intrapericardial approach was adopted for esophageal dissection.
Upon entering the pericardium, it was evident that a single fibrotic membrane had replaced the left atrial wall, the pericardium, and the anterior esophageal wall. Moreover, a small red bleb within an esophageal sinus observed during preoperative gastroscopy in fact represented an esophageal-left atrial fistula covered by a tenuous thin membrane of fibrin. During dissection, this 2 x 2 mm defect began to bleed. It was controlled with a pursestring 4-0 polypropylene suture (Fig 3). The esophagus was divided immediately proximal to the azygous vein and the surgical specimen comprising the distal two thirds of the esophagus and proximal stomach was removed. The previously prepared gastric conduit was brought into the right chest through an enlarged esophageal hiatus and was used to buttress the left atrial repair.
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Fig 3. After repair of the esophageal-left atrial fistula, the left atrium is being retracted to expose the 4-0 polypropylene pursestring suture used to exclude the weakened atrial wall. (LA = left atrium; LMB = left main bronchus; RLL = right lower lobe; RMB = right main bronchus; white arrow = pericardial edge; white arrowhead = 4-0 polypropylene pursestring suture excluding the weakened atrial wall.)
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Restoration of intestinal continuity was completed with a supra-azygous single layer hand sewn esophagogastric anastomosis. The patient had an uneventful postoperative course and was discharged on day 13 tolerating a full diet. Final pathology revealed a distal esophagus completely replaced by radiation-induced fibrosis and no evidence of malignancy. The patient is currently 8 months postresection with no further bleeding or dysphagia and no evidence of recurrence.
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Comment
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Nontraumatic esophageal-atrial fistula is a rare condition that is almost invariably fatal. Survival for this entity is extremely rare. We believe that this is the second case that has been successfully treated, and the first case involving the left atrium. The mechanism of fistula formation in our patient differs from that of patients suffering from the complications of radiofrequency ablation for atrial fibrillation. Indeed, she had several well-recognized risk factors for fistula formation including malignancy, previous radiotherapy, and multiple esophageal dilations, present in respectively 17%, 60%, and 11% of the cases reviewed by Snyder and colleagues [1].
Early diagnosis of this entity is challenging due to its low incidence, nonspecific early symptoms, and failure of noninvasive diagnostic tools. Patients with subsequently identified esophageal-atrial fistulas rarely have sentinel warning signs other than gastrointestinal bleeding [2]. Although in the present case the presence of the fistula was not recognized on preoperative imaging, in retrospect, the ulcer and sinus on endoscopy both represented signs suggestive of the fistula.
Death in all previous cases has resulted from movement of blood, air, or food across the fistula with ensuing hemorrhage or neurologic deficits incompatible with life [3]. In the present case, a contained communication between the atrium and esophagus had developed over several months and was finally opened with instrumentation (dilation and biopsy) at endoscopy.
Management of an esophageal-atrial fistula is challenging and requires prompt intervention. As this case demonstrated, the Sengstaken-Blakemore tube (Rüsch), designed for controlling variceal bleeding, is a useful, if temporary, adjunct to the management of this difficult problem. Definitive treatment usually necessitates a direct surgical approach with resection of the diseased esophagus, closure of the atrial defect, and buttressing the repair with nonirradiated tissue (ie, in this case the stomach) to prevent recurrent bleeding. In poor surgical candidates due to prohibitive comorbidity or diffuse metastatic disease, placement of a self-expanding metallic stent represents a viable option [4].
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References
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- Snyder RW, Dumas PR, Kolts BE. Esophageal fistula with previous pericarditis complicating esophageal ulcerationReport of two cases and review of the literature. Chest 1990;98:679-681.[Medline]
- Cummings JE, Schweikert RA, Saliba WI, et al. Brief communication: atrial-esophageal fistulas after radiofrequency ablation Ann Intern Med 2006;144:572-574.[Abstract/Free Full Text]
- Reynolds P, Walker FO, Eades J, Smith JD, Lantz PE. Food embolus J Neurol Sci 1997;149:185-190.[Medline]
- 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]
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Abstract
Introduction
