Surg Today DOI 10.1007/s00595-013-0744-9

CASE REPORT

Successful non-operative management of left atrioesophageal fistula following catheter ablation Manabu Shiraishi • Hideki Morita • Kenichi Muramatsu • Akira Sato • Junichi Nitta Atsushi Yamaguchi • Hideo Adachi



Received: 26 November 2012 / Accepted: 25 February 2013 Ó Springer Japan 2013

Abstract Atrioesophageal fistula (AEF) is a potentially lethal complication of catheter radiofrequency ablation for atrial fibrillation. A 49-year-old man with paroxysmal atrial fibrillation who underwent catheter ablation around the pulmonary vein was admitted 31 days after the procedure, suffering seizures and fever. Magnetic resonance imaging of the brain showed ischemia and multiple lesions of acute infarction in the right occipital lobe of the cerebrum. Computed tomography (CT) of the chest showed a small accumulation of air between the posterior left atrium and the esophagus, suggesting an AEF. Endoscopic snaring of the esophageal mucosa, repeated a few times, supported by nil by mouth and antibiotic therapy, resulted in improvement of his condition with no recurrence of symptoms. Subsequent chest CT scans confirmed disappearance of the leaked air and the patient was discharged home 45 days after admission with no neurological compromise.

Introduction

Keywords Atrioesophageal fistula  Catheter ablation  Radiofrequency ablation

Case report

M. Shiraishi (&)  H. Morita Division of Cardiovascular Surgery, Saitama Red-cross Hospital, 8-3-33 Kamiochiai, Chyuo-ku, Saitama, Saitama 338-8553, Japan e-mail: [email protected] K. Muramatsu  A. Sato  J. Nitta Division of Cardiovascular Internal Medicine, Saitama Red-cross Hospital, Saitama, Japan A. Yamaguchi  H. Adachi Department of Cardiovascular Surgery, Saitama Medical Center, Jichi Medical University, Saitama, Japan

Catheter radiofrequency ablation (RFA) for atrial fibrillation is now well established as a treatment strategy for medication-refractory symptomatic atrial fibrillation [1]. However, it does carry a risk of the rare but lethal complication of atrioesophageal fistula (AEF), associated with significant neurological mortality and high morbidity. Despite general agreement that prompt surgical repair is critical for the survival of such patients, the optimal approach has not been established. Many cases of successful surgical repair of AEF have been reported, but successful conservative management has rarely been described. We report a case of AEF after RFA for atrial fibrillation, which was treated successfully without major surgery.

A 49-year-old man with a 6-year history of paroxysmal atrial fibrillation, which was highly symptomatic despite antiarrhythmic drugs, underwent RFA around the pulmonary vein and was discharged uneventfully in sinus rhythm. However, he was readmitted 31 days later after the sudden onset of fever, followed by left hemiplegia and seizures. On admission, the patient was hypotensive, with a fever of 40 °C, and ECG showed atrial fibrillation with a rapid ventricular response of 180 bpm. Laboratory data were notable for an elevated C-reactive protein level of 6.6 mg/dl. Magnetic resonance imaging (MRI) scans of the brain revealed multiple areas of acute cerebral infarction in the right occipital lobe of the cerebrum (Fig. 1). Initially, we suspected infective endocarditis or bacterial cerebral meningitis as the cause of the cerebral

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Surg Today Fig. 1 Cerebral magnetic resonance imaging showed diffuse ischemic changes consistent with multiple emboli (arrow)

Fig. 2 Computed tomogram showed an air pocket within the esophageal wall and the posterior wall of the left atrium (arrow)

infarction. Blood cultures were positive for gram-positive coccus, but transesophageal echocardiography showed no evidence of endocarditis. Cerebrospinal fluid analysis was also unremarkable. Intravenous antibiotic therapy was initiated and the patient was observed for several days. The fever subsided completely within 48 h after admission, but he continued to have recurring episodes of hemiplegia, seizures and transient ST-segment elevation on ECG while eating, with complete resolution within a few hours. A computed tomography (CT) scan of the chest disclosed a small accumulation of air in the region of the left atrium adjacent to the esophagus (Fig. 2), suggesting an AEF. The gram-positive coccus on blood culture might have been the normal flora of the mouth. A gastric tube was placed and the patient was kept on nil by mouth. Gastroesophageal endoscopy showed a pinhole lesion and scar in the esophagus with no signs of acute inflammation or bleeding (Fig. 3). We snared the esophageal mucosa to close the pinhole lesion. To ensure that there was no communication between the esophagus and the left atrium, the patient was given a fluoroscopic series of ion-containing contrast agent swallows, which showed no evidence of residual

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Fig. 3 Esophagoscopy 40 days after radiofrequency ablation (RFA) showed a pinhole lesion and scar in the esophagus with no signs of acute inflammation or bleeding

fistula. We selected conservative treatment because the early intravenous antibiotics with nothing by mouth had improved the bacteremia and symptoms of air embolism.

Surg Today

A chest CT scan done to re-evaluate the AEF, 2 weeks after endoscopic repair, showed no pneumomediastinum. Thus, the patient was allowed sips of clear fluids and advanced to free fluids and finally to a soft solid diet, while monitoring his clinical status, which remained stable. Laboratory data related to inflammation improved and he was discharged home 45 days after admission. He remained free of any signs of recurrence of symptoms in his 6-month follow-up. He was able to grade up to a regular diet and has not suffered any recurrent neurological compromise or required further antibiotic treatment.

Discussion RFA of the atrial wall is becoming an increasingly common treatment for arrhythmias. Collateral damage from the energy source is a well-identified complication and associated injury to the esophagus has been documented [1–8]. RFA-induced esophageal fistula has been universally associated with significant morbidity and, if not recognized and treated promptly, serious disability or death usually ensue [2]. Death is usually imminent [4, 7, 8], although survival is possible with prompt diagnosis and emergency surgery [1, 3, 5, 6]. Mild thermal injury to the esophagus occurs in up to 2.9 % of patients, but an AEF develops in only 0.05–1 % of patients undergoing RFA [1, 4]. Although rare, AEF is often fatal with a mortality rate greater than 70 % [1]. Symptoms can manifest anywhere from 3 days to as late as in 41 days after ablation [4]. Early symptoms and signs, although not specific, are reflective of an inflammatory process in the mediastinum, endocarditis or pericarditis and may include chest pain, fever, melena, hematemesis, arrhythmias, and gastrointestinal bleeding. Symptoms can also be caused by polymicrobial sepsis and cerebral air embolism and are usually fatal events [2]. Our patient presented 31 days after ablation, with fever, hemiplegia, and seizures. He had no history of stroke prior to RFA, and this was the first episode of atrial fibrillation and cerebral infarction after RFA. The deterioration of his general condition and the high fever might have triggered the recurrence of atrial fibrillation and hypotension. Thus, AEF should be considered in any patient presenting with a high fever, neurologic symptoms, or bleeding after RFA to initiate prompt treatment [6]. Two possible mechanisms have been proposed to explain the development of an AEF: One involves injury to the esophageal vasculature, leading to an ischemic lesion [9]. The other involves the initial thermal injury followed by progressive lesion enlargement promoted by the reflux of stomach acid bathing the lesion [10]. Our patient suffered the sudden onset of recurring episodes of hemiplegia

and seizures, as well as transient ST-segment elevation while eating. One report hypothesizes that air emboli can develop as a result of the esophagus being able to produce pressure 10 times greater than intra-atrial pressure [11]. With this pressure gradient, air is sucked into the left atrium from the esophagus, causing embolisms in the cerebral blood vessels and coronary arteries. Preventing the process that leads to an AEF is the most important measure. Since experiencing this case, we routinely check the positional relationship between the left atrium and esophagus before RFA and measure the temperature of the esophageal mucosa during RFA to prevent AEF. We set a higher limit to stop it exceeding 42 °C. Ultimately, oral care may be the most important preventive measure. If AEF is suspected at all, chest CT is the most useful diagnostic modality as it specifically shows air within or adjacent to the left atrium. Brain MRI can also demonstrate evidence of embolic infarct. We used gastroesophageal endoscopy to expose and treat the AEF; however, gastroesophageal endoscopy and transesophageal echocardiography have been reported to enlarge the fistula tract and aggravate air embolization. Thus, these procedures are often considered contraindicated [1, 3]. For this reason, endoscopic treatment that increases pressure in the esophagus is not recommended in the early stage. Besides, damaged esophageal mucosa may be very weak and predisposed to rupture in a patient with early onset AEF following RFA. In the later stage, it might be sufficient to observe the esophageal mucosa, which can heal with scar formation. We decided that this lesion was better treated conservatively than surgically after the patient responded so well to intravenous antibiotics supported by nil by mouth, with improved bacteremia and resolution of the symptoms of air embolism. Fortunately, the thermal injury was very small and the scarring with successful endoscopic fistula closure treatment saved our patient. This complication is associated with very high morbidity and mortality and urgent surgical repair is the standard treatment [4]. Several authors have reported that early recognition and prompt surgical intervention averted a fatal outcome [1, 3, 5, 6]. The surgical approaches to repair the AEF include left thoracotomy and right posterolateral thoracotomy [6]. If active bleeding continues under beating or fibrillation, these approaches allow us to block off the bleeding with cardioplegic arrest by aortic cross clamping. After primary repair of the AEF, maintaining physical separation between the esophagus and atrium with a muscle flap or pericardial patch is crucial to prevent re-fistulization [6]. Another technique of surgical repair was described in a single case of successful esophageal stenting [12]. However, this approach entails the attendant risk of air embolism during insufflation. Regardless of the surgical

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technique, immediate initiation of empirical antimicrobial therapy with high blood–brain barrier permeability is strongly advised for patients with a suspected AEF [5]. Our patient suffered the initial neurologic deficit as late as 31 days after the procedure. The primary differential diagnoses on admission were infective endocarditis or meningitis, so empirical antibiotics were instituted. The subsequent definitive diagnosis was based on the clinical presentation and the chest CT finding of air behind the left atrium. Whereas most cases of AEF are treated by emergency surgical intervention [1, 3–8], we chose conservative therapy, which proved successful in this case. We recommend starting intravenous antibiotics treatment as soon as possible for preventing mediastinitis, endocarditis or pericarditis. Moreover, patients should be kept on nil by mouth to prevent an air embolism. If infection cannot be controlled with intravenous antibiotics treatment, surgery should be performed without further delay. In summary, we described the successful non-surgical management of an AEF after RFA for AF. In a patient with high index of suspicion for AEF after RFA, the chest CT finding of a small accumulation of air in the region of the left atrium adjacent to the esophagus is the most useful modality to confirm an early diagnosis. This case demonstrates that conservative treatment is a potential option for AEF and that prompt empirical initiation of antibiotics is crucial for survival. Acknowledgments presented.

No external funding was obtained for the work

Conflict of interest

We have no conflicts of interest to declare.

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Successful non-operative management of left atrioesophageal fistula following catheter ablation.

Atrioesophageal fistula (AEF) is a potentially lethal complication of catheter radiofrequency ablation for atrial fibrillation. A 49-year-old man with...
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