[Double aortic arch and aortotracheal fistula as cause of fatal massive bleeding: a rare and dangerous association]. / Doble arco aórtico y fistula aortotraqueal como causa de hemorragia masiva fatal: una asociación rara y peligrosa.

Vascular rings are unusual congenital malformations. Among them, double aortic arch (DAA) is often difficult to diagnose due to its low incidence of symptoms. DAA can be associated with tracheal or esophageal compression and, in severe cases, could require tracheal intubation or chronic use of a nasogastric tube. This scenario favors the development of aortotracheal fistulas (ATF) or aortoe-sophageal fistulas (AEF). OBJECTIVE: To present a clinical case with an unusual association of DAA with ATF and to reinforce the importance of maintaining high diagnostic suspicion in patients with massive aerodigestive bleeding without an obvious source. CLINICAL CASE: A 32-week preterm newborn who required prolonged mechanical ventilation and presented intermittent episodes of massive oropharyngeal bleeding with hemodynamic compromise associated with lower airway obstruction without pulmonary hemorrhage. The patient underwent upper endoscopy and exploratory laparotomy without evidence of bleeding. Flexible nasopharyngolaryngoscopy and direct laryngoscopy also showed no abnormalities. A CT angiography showed complete DAA with indentation of the left dominant arch over the trachea, without severe stenosis or evidence of a fistula. AEF was suspected, so exploratory surgery was considered. However, the patient died before surgery due to a massive pulmonary hemorrhage. The autopsy revealed the presence of ATF. CONCLUSIONS: In patients with massive aerodigestive bleeding without an obvious source, the presence of DAA and possible AEF/ ATF should be considered. Imaging studies have a poor performance for this diagnosis, so surgery should be considered for diagnosis and treatment in these patients.

Impact of pulsed field ablation on intraluminal esophageal temperature.

INTRODUCTION: Atrio-esophageal fistula after esophageal thermal injury (ETI) is one of the most devastating complications of available energy sources for atrial fibrillation (AF) ablation. Pulsed field ablation (PFA) uses electroporation as a new energy source for catheter ablation with promising periprocedural safety advantages over existing methods due to its unique myocardial tissue sensitivity. In preclinical animal studies, a dose-dependent esophageal temperature rise has been reported. In the TESO-PFA registry intraluminal esophageal temperature (TESO) changes in a clinical setting are evaluated. METHODS: Consecutive symptomatic AF patients (62 years, 67% male, 61% paroxysmal AF, CHA2 DS2 Vasc Score 2) underwent first-time PFA and were prospectively enrolled into our registry. Eight pulse trains (2 kV/2.5 s, bipolar, biphasic, x4 basket/flower configuration each) were delivered to each pulmonary vein (PV). Two extra pulse trains per PV in flower configuration were added for wide antral circumferential ablation. Continuous intraluminal esophageal temperature (TESO) was monitored with a 12-pole temperature probe. RESULTS: Median TESO change was statistically significant and increased by 0.8 ± 0.6°C, p < .001. A TESO increase ≥ 1°C was observed in 10/43 (23%) patients. The highest TESO measured was 40.3°C. The largest TESO difference (∆TESO) was 3.7°C. All patients remained asymptomatic considering possible ETI. No atrio-esophageal fistula was reported on follow-up. CONCLUSION: A small but significant intraluminal esophageal temperature rise can be observed in most patients during PFA. TESO rise over 40°C is rare. The clinical implications of the observed findings need to be further evaluated.

Fatal cerebral air embolism from atrio-oesophageal fistula following cardiac ablation.

A young woman with Rogers syndrome (thiamine-responsive megaloblastic anaemia, diabetes mellitus and sensorineural deafness) presented with headache, recurrent supraventricular tachycardia and features of an upper gastrointestinal bleed, 1 month after radiofrequency cardiac ablation for supraventricular tachycardia. She deteriorated rapidly after endoscopy and subsequently died. Brain imaging during the acute deterioration showed diffuse intracranial air embolism and hypoxic-ischaemic injury. Postmortem examination showed an atrio-oesophageal fistula, a rare complication of cardiac ablation. Clinicians should suspect this condition in patients with acute neurological deterioration after cardiac ablation who have diffuse air embolism on imaging.

[Beating heart surgical treatment of atrio-esophageal fistula after radiofrequency ablation of atrial fibrillation]. / Trattamento chirurgico a cuore battente di una fistola atrio-esofagea post-ablazione tramite radiofrequenza di fibrillazione atriale.

Atrio-esophageal fistula (AEF) is a rare (0.02-0.1%) complication of radiofrequency ablation for atrial fibrillation and is associated with high mortality. It typically presents between 2 and 6 weeks after catheter ablation. AEF was reported to be the second complication as cause of death after radiofrequency ablation with a mortality rate of 71%. Common clinical features of AEF include dysphagia, nausea, heartburn, hematemesis or melena, high fever, sepsis, pericardial or pleural effusions, mediastinitis, seizures, and stroke. Once the diagnosis of AEF is made, early surgical repair is mandatory. Herein, we report a case of a AEF treated surgically without extracorporeal circulation.

Surviving the nonsurvivable combination of a mycotic aneurysm progressing into a concomitant aorto-bronchial- and aorto-esophageal fistula, a case report.

BACKGROUND: Aortic mycotic aneurysms are a rare but life-threatening condition and may be associated with aorto-bronchial- and aorto-esophageal fistulas. Although both very rare, they carry a high mortality and require (urgent) surgical intervention. Surviving all three conditions concomitantly is extraordinary. We describe a patient who underwent staged repair of such combined defects.

Chicken Bone Ingestion Leads to Aortoesophageal Fistula With Catastrophic Bleeding.

Aortoesophageal fistula (AEF) is an uncommon, but potentially fatal cause of upper gastrointestinal bleeding. Aortoesophageal fistulas caused by foreign body ingestion are rare but devastating. The classic clinical triad of AEF consists of mid-thoracic pain or dysphagia, a herald episode of hematemesis, followed by fatal exsanguination after a symptom-free period (Chiari's triad). Computed tomography angiography (CTA) is the preferred diagnostic tool for identifying AEF and is substantially more sensitive than upper endoscopy for detecting AEF. Endoscopy can detect AEF as it might show pulsatile blood, pulsatile mass, hematoma, or adherent blood clot in the esophagus, or a deep esophageal tear. However, endoscopy has a low sensitivity and may delay definitive treatment. Several management options for AEF have been suggested; however, the definitive treatment is surgery performed on the thoracic aorta and esophagus, including esophagectomy, surgical replacement of the thoracic aorta, thoracic endovascular aortic repair, or omental flap. We report a case of a 63-year-old man who presented with hematemesis 2 weeks after chicken bone ingestion.

Esophago-Vascular Fistulae in Children: Five Survivors, Literature Review, and Proposal for Management.

INTRODUCTION: Esophago-vascular fistulae in children are almost uniformly fatal with death occurring by exsanguination. We present a single centre series of five surviving patients, a proposal for management and literature review. MATERIALS AND METHODS: Patients were identified from surgical logbooks, surgeon recollection and discharge coding data. Demographics, symptoms, co-morbidities, radiology, management and follow up details were recorded. RESULTS: Five patients (1M, 4F) were identified. Four were aorto-esophageal and one caroto-esophageal. Median age at initial presentation was 44 (8-177) months. Four patients had cross sectional imaging prior to surgery. Median time from presentation to combined entero-vascular surgery was 15 (0-419) days. Four patients required repair on cardio-pulmonary bypass with four undergoing staged surgical procedures. All required combined esophageal and cardio-vascular surgery. Length of PICU stay following combined surgery was 4 (2-60) days and overall hospital stay was 53 (15-84) days. Median follow up was 51 (17-61) months. Two patients had esophageal atresia and trachea-esophageal fistula managed as neonates. Three had no co-morbidities. Four had esophageal foreign bodies:1 esophageal stent, 2 button batteries, 1 chicken bone. One patient had a complication following colonic interposition. Four patients required an esophagostomy at the time of definitive surgery. All patients were alive and well at last follow up with one having successful reconnection surgery. CONCLUSION: In this series, outcomes were favourable. Multidisciplinary discussion and surgery are mandatory. If hemorrhage is controlled at presentation, then survival to discharge is possible but the magnitude of surgical intervention is both significant and very high risk. LEVEL OF EVIDENCE: Level 3.

Surgical Treatment of Atrioesophageal Fistula: A Systematic Review.

BACKGROUND: Atrioesophageal fistula is a rare and morbid complication of ablation therapy for atrial fibrillation. Surgery provides increased survival; however, which surgical approach provides the best outcome is unclear. METHODS: We performed a retrospective analysis of cases in the literature and at our institution. We characterized patients by presenting symptoms, diagnostic method, surgical therapy with different approaches, and survival. RESULTS: In total, 219 patients were found, with 216 patients identified from 122 papers in the literature and 3 patients from our institutional database (2000-2022). The most common presenting symptoms included fever/chill (71.8%) and neurologic deficiency (62.9%). The overall survival for this cohort was 47%. Patients who had an operation had significantly improved survival compared with those who did not have an operation (71.9.3% vs 11%, P < .001). Patients who survived after surgical intervention typically underwent right thoracotomy (45.1%), patch repair of the left atrium (61.1%), and primary repair of the esophagus (68.3%) on cardiopulmonary bypass (84.8%) with a flap between the 2 organs (84.6%). Patients who had cardiopulmonary bypass had increased survival (39 of 45 [86.7%]) compared with those who did not have cardiopulmonary bypass (7 of 17 [41.2%], P < .001). CONCLUSIONS: Patients with atrioesophageal fistula should undergo surgical intervention. A patch repair of the left atrium and primary repair of the esophagus with a flap between the organs during cardiopulmonary bypass is the most common successful repair. Cardiopulmonary bypass may allow better débridement and repair of the left atrium, which may provide a survival advantage in the treatment of this rare disease.

A worldwide survey on incidence, management, and prognosis of oesophageal fistula formation following atrial fibrillation catheter ablation: the POTTER-AF study.

AIMS: Oesophageal fistula represents a rare but dreadful complication of atrial fibrillation catheter ablation. Data on its incidence, management, and outcome are sparse. METHODS AND RESULTS: This international multicentre registry investigates the characteristics of oesophageal fistulae after treatment of atrial fibrillation by catheter ablation. A total of 553 729 catheter ablation procedures (radiofrequency: 62.9%, cryoballoon: 36.2%, other modalities: 0.9%) were performed, at 214 centres in 35 countries. In 78 centres 138 patients [0.025%, radiofrequency: 0.038%, cryoballoon: 0.0015% (P < 0.0001)] were diagnosed with an oesophageal fistula. Peri-procedural data were available for 118 patients (85.5%). Following catheter ablation, the median time to symptoms and the median time to diagnosis were 18 (7.75, 25; range: 0-60) days and 21 (15, 29.5; range: 2-63) days, respectively. The median time from symptom onset to oesophageal fistula diagnosis was 3 (1, 9; range: 0-42) days. The most common initial symptom was fever (59.3%). The diagnosis was established by chest computed tomography in 80.2% of patients. Oesophageal surgery was performed in 47.4% and direct endoscopic treatment in 19.8% and conservative treatment in 32.8% of patients. The overall mortality was 65.8%. Mortality following surgical (51.9%) or endoscopic treatment (56.5%) was significantly lower as compared to conservative management (89.5%) [odds ratio 7.463 (2.414, 23.072) P < 0.001]. CONCLUSION: Oesophageal fistula after catheter ablation of atrial fibrillation is rare and occurs mostly with the use of radiofrequency energy rather than cryoenergy. Mortality without surgical or endoscopic intervention is exceedingly high.

[Aortoesophageal Fistula Induced by an Indwelling Nasogastric Tube: A Case Report].

A 91-year-old woman who presented with melena and hypovolemic shock visited the emergency room. She received enteral nutrition by nasogastric tube in a bedridden state due to hip surgery. Gastroscopy initially suggested a simple ulcer that occurred after a nasogastric tube was placed for a long time, but the ulcer was deep, and the amount of instantaneous bleeding was considerable. Therefore, an aortoesophageal fistula was suspected. Angiography was performed instead of endoscopic hemostasis, followed by thoracic endovascular aortic repair (TEVAR). After the TEVAR procedure, the patient recovered without further gastrointestinal bleeding. Prompt judgment and communication between the endoscopist and the interventional physician are important for successful hemostasis in an aortoenteric fistula patient.

Acquired atrioesophageal fistula: Need it be lethal? Sizing up the problem, diagnostic modalities, and best management.

BACKGROUND AND AIM OF THE STUDY: An atrioesophageal fistula is a devastating complication of ablation for atrial fibrillation. For the surgeon facing this dreaded complication, it may be a 'once in a lifetime' case. This review aims to describe the clinical problem and evaluate the outcome of different surgical techniques to start guiding cardiothoracic surgeons toward those which offer the best chance of survival. METHODS: An electronic search retrieved 125 articles containing 195 cases of atrioesophageal fistula secondary to atrial fibrillation ablation. Reports of pericardio-esophageal or mediastino-esophageal fistula were excluded. RESULTS: The median age was 61 and 143 (73%) cases occurred in males. Fever (n = 147; 75%) and neurological dysfunction (n = 151; 77%) were the most common symptoms. The median time from ablation to symptom onset was 21 days (interquartile range: 12-28). The most sensitive thoracic imaging modality was computed tomography (n = 135/153; 90%). Immediate deterioration occurred during 11/58 (19%) oesophago-gastro-duodenoscopies. Mortality was lower in patients who had surgery (39%) compared with endoscopic intervention (94%) or conservative management (97%). Patients who had atrial repair combined with esophageal repair or oesophagectomy were more likely to survive than those who had atrial repair alone (OR 6.97; p < .001). Isolation of the esophageal aspect of the fistula conferred an additional survival benefit (OR 5.85; p = .02). CONCLUSIONS: Fever, neurological symptoms, and chest pain in the context of recent ablation should prompt immediate evaluation. Urgent CT thorax should be arranged and repeated if initially unremarkable. Esophageal instrumentation should be avoided due to the risk of catastrophic air embolism or massive hemorrhage. The best way forward is emergency surgical repair; the combination which offers the best survival benefit is atrial repair combined with esophageal surgery and isolation of the esophageal aspect of the fistula.

Esophageal Left Subclavian Artery Fistula Caused by The Esophageal Foreign Body: A Case Report.

Left subclavian artery esophageal fistula usually occurs after esophageal cancer surgery, which is a rare complication, and it is even rarer after stent implantation of left subclavian artery pseudo-aneurysm. This paper reports the case of a 21-year-old male patient with left subclavian artery pseudo-aneurysm. Two-plus months after stent implantation, he stopped anticoagulant and antiplatelet drugs and developed pain in his left upper limb. The patient was diagnosed with arterial fistula. He was discharged from the hospital successfully after several operations, such as thoracic aortic stent implantation, left common carotid artery left axillary artery artificial vascular bypass. Conclusion: Early diagnosis and positive treatment lead to a good prognosis for patients with esophageal left subclavian artery fistula.

Tubercular Bronchoesophageal Fistula in an Adolescent Girl.

Bronchoesophageal fistula is a rare complication of Mycobacterium tuberculosis in children. An adolescent girl who was diagnosed of tubercular mediastinal lymphadenopathy with associated bronchoesophageal fistula at presentation, is reported here. This 16-y-old girl presented with high-grade fever, cough, decreased appetite, weight loss for 3 mo, and breathlessness for 10 d. Chest radiograph revealed hilar lymphadenopathy with bilateral pleural effusion. GA GeneXpert was positive for mycobacterium and rifampicin sensitivity. Despite antitubercular therapy cough persisted and there was a history of dry cough with food intake, especially more on liquids. Bronchoscopy and CECT chest confirmed bronchoesophageal fistula in the right main bronchus just below the carina. Child continued on tube feeding and antitubercular therapy. After completion of intensive phase, child improved with resolution of clinical symptoms and scarring of tract on repeat bronchoscopy. It is concluded that in children with combination of mediastinal lymphadenopathy and persistent cough following intake of food needs careful evaluation for trachea/bronchoesophageal fistula.

Delayed aortoesophageal and tracheoesophageal fistulas secondary to foreign body ingestion: a case report.

Ingestion of a foreign body (FB) is a common condition with a few potentially life-threatening complications, including esophageal perforation (EP), aortoesophageal fistula (AEF), mediastinal infection, and tracheoesophageal fistula (TEF). In this case, a patient who accidentally ingested a duck bone gradually experienced all of the above complications. To resolve the symptom of difficulty swallowing, the patient underwent emergency treatment for removal of the esophageal FB via endoscopic surgery. Under endoscopy, esophageal mucosal injuries were present, but no other abnormalities, such as active bleeding, were observed. However, the patient returned to our hospital a week later with symptoms of vomiting and black stool and received the diagnosis of EP, AEF and mediastinal infection. Two days later, he vomited 1,000-2,000 mL of blood after experiencing sudden severe chest pain. Then, thoracic endovascular aortic repair (TEVAR) and mediastinal drainage with video-assisted thoracoscopic surgery (VATS) were performed under emergency general anesthesia. Additionally, the patient underwent esophageal stent implantation when TEF was confirmed by tracheal computed tomography (CT). The patient was treated with anti-infective therapy throughout the treatment process. Finally, he recovered and was able to tolerate a liquid diet. Comprehensive evaluation and multidisciplinary cooperation are all very important for the treatment of esophageal foreign bodies and complications.

Autopsy Techniques in Patients With History of Radiofrequency Ablation for Atrial Fibrillation.

CONTEXT.­: Esophageal fistula formation is one of the most feared complications of radiofrequency catheter ablation. This procedure and its many variations, such as the "maze," are becoming the mainstream treatment for atrial fibrillation owing to limitations of antiarrhythmic drugs. The incidence of this complication rate has been reported to be from 0.01% to 1%. OBJECTIVE.­: To delineate the importance of using the en bloc Letulle method of dissection for identifying esophageal fistulas for cases with a history of radiofrequency catheter ablation. DESIGN.­: Six autopsy cases with a history of radiofrequency catheter ablation for atrial fibrillation were selected from 1736 autopsies performed between 2009 and 2020. RESULTS.­: The initial presenting symptoms included neurologic symptoms, chest pains, epigastric discomfort, and sepsis. Transesophageal echocardiogram in 4 cases showed no evidence of thrombus or vegetation, however, 2 cases had evidence of atrial esophageal fistula. The autopsy findings included 5 atrial esophageal fistulas and 1 esophagopericardial fistula. Atrial esophageal fistulas were small and could be detected without difficulty when the en bloc Letulle technique was used and would have been easily missed by the Virchow method. The immediate causes of the deaths were myocardial ischemia, septic emboli to brain and heart, hypovolemic shock secondary to exsanguination, stroke, and coagulopathy. CONCLUSIONS.­: To date, this is the largest collection of autopsy cases showing esophageal fistula associated with prior radiofrequency catheter ablation. The Letulle dissection method is preferable in this setting.

Optimal Surgical Approach to Left Atrioesophageal Fistula After Catheter Radiofrequency Ablation.

Atrioesophageal fistula formation is a rare but formidable complication after catheter radiofrequency ablation for atrial fibrillation. We present 4 patients who underwent urgent primary intracardiac repair of the left atrium via sternotomy with central cardiopulmonary bypass and early aortic cross-clamp, followed by repair of the esophagus. We believe that this approach represents the safest strategy for addressing this morbid and often fatal complication.

Clinical Manifestations, Outcomes, and Mortality Risk Factors of Atrial-Esophageal Fistula: A Systematic Review.

INTRODUCTION: Atrial-esophageal fistula (AEF) is a rare but life-threatening complication of catheter ablation. The clinical presentation and mortality risk factors of AEF have not been fully elucidated. The aim of this study was to systematically review the clinical characteristics and prognosis of AEF. METHODS: PubMed was searched from inception to October 2020 following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses statement protocol. RESULTS: A total of 190 AEF patients were included. The mean age was 59.29 ± 11.67 years, 74.21% occurred in males, and 81.58% underwent radiofrequency ablation. AEF occurred within 30 days after ablation in 80.82% of patients and occurred later in patients presenting with neurological symptoms compared with other symptoms (median of onset time: 27.5 days vs. 16 days, p < 0.001). Clinical presentation included fever (81.58%) and neurological symptoms (80.53%). Chest computed tomography (abnormal rate of 91.24%) was the preferred diagnostic test, followed by magnetic resonance imaging of the brain (abnormal rate of 90.91%). Repeated testing improved diagnostic evaluation sensitivity. Distinctive imaging results included free air in the mediastinum (incidence rate of 81.73%) and air embolism of the brain (incidence rate of 57.53%). The overall mortality was 63.16%, with worse nonsurgical treatment outcomes compared with outcomes of surgical treatment (94.19% vs. 33.71%, p < 0.001). Conservative or stent intervention was an independent risk factor for mortality. Age (adjusted odds ratio, 1.063, p = 0.004), presentation with neurological symptoms (adjusted odds ratio, 5.706, p = 0.017), and presentation with gastrointestinal bleeds (adjusted odds ratio, 3.009, p = 0.045) were also predictors of mortality. CONCLUSIONS: AEF is a fatal ablation complication. AEF can be diagnosed using a combination of a clinical history of ablation, infection, or neurological symptoms and an abnormal chest CT. Our analysis supports that surgical treatment reduces the mortality rate.