Abstract
The carotid-esophageal fistula is a rare and serious complication of the metallic esophageal prosthesis. A high index of suspicion is required for early diagnosis and treatment, decreasing the morbidity and mortality rate of this severe complication. We report a case of a 4-year-old boy presenting severe upper gastrointestinal bleeding due to a carotid-esophageal fistula, secondary to deployment of an esophageal metallic prosthesis for treatment of a recurrent stenosis. The carotid pseudo-aneurism was successfully treated with stents and coils. Although endovascular treatment is a safe and effective option, arterial stenting in children needs further studies with long-term follow-up.
Introduction
Esophageal atresia is the most common congenital malformation affecting the esophagus, with an incidence of 1 per 2,500-4,500 live-births. 1 The treatment is predominantly surgical, and anastomosis stenosis is one of the most frequent postoperative complications. 1 Even though most cases respond to one or 2 endoscopic dilations, 1,2 some patients require alternative treatment, including intralesional corticosteroid injection, placement of metallic stents and plastic esophageal prosthesis. 2 Complications associated with stents include epithelialization, pain, reflux, stent migration, esophageal perforation and fistula with adjacent structures, which rarely involves the common carotid artery. 2,3 We further report a case of a boy with recurrent esophageal stenosis and esophageal metallic stent, complicated with a carotid-esophageal fistula (CEF) successfully treated through endovascular approach.
Case Report
A 4-year-old Down syndrome boy with was diagnosed at birth with long-gap esophageal atresia, being submitted to esophagostomy and gastrostomy. He later underwent a difficult end-to-end esophageal anastomosis, developing postoperative stricture. It was treated with by utilizing endoscopic stenotomy with intralesional injection of corticosteroids and several endoscopic dilations, one of them complicated by pneumomediastinum. The esophageal anastomotic stricture was also refractory to metallic stent placement (which migrated), magnetic anastomosis and a redo esophagoplasty through thoracotomy and end-to-end anastomosis. Two years later, after new endoscopic dilations, a new metallic stent was placed (Figure 1A). Just after a complete treatment for pneumonia, with piperacillin/tazobactan, the patient presented hematemesis and melena, and the migrated stent was endoscopically repositioned. During the hospitalization, the patient had 2 more massive episodes of upper gastrointestinal bleeding, and a new endoscopy showed no significant findings. No clinical or radiological signs of infection were found. However, the patient developed shock and dyspnea, managed with mechanic ventilation, fluid resuscitation and transfusion. Vancomycin and meropenem were introduced, as guided by the Hospital Infection Control Committee. After stabilization, CT-scan evidenced a hemothorax, and a CT-angiography suggested lesion in the right common carotid artery (CCA) (Figure 1B). Thus, a digital subtraction angiography (DSA) confirmed a 4 mm saccular pseudoaneurysm in proximal segment of the right CCA, which had bled into the esophagus and thoracic cavity. Embolization was tried through a microcatheter (Excelsior SL-10; Boston Scientific, Natick, MA, USA) passing in through a 5F guiding-catheter (Guider Softip; Boston Scientific) over a microguidewire (Transcend; Boston Scientific); however, the wide aneurysmal neck did not allow safe deployment of coils. Hence, the introducer was changed for a 6F sheath, and a 5 × 26 mm stent [PRO-Kinetic Energy, Biotronic AG, Bülach, Switzerland] was implanted in the CCA, being followed by aneurysm occlusion with deployment of 4 detachable platinum coils (Barricade; Blockade Medical, Irvine, CA, USA) (Figure 2A-C). Postoperative period in Intensive Care Unit was uneventful, and acetylsalicylic acid (ASA) was administered and maintained at 85 mg per day. The esophageal stent was endoscopically removed on the 7th postoperative day (Figure 1C), and a new hemorrhage occurred 1 month later. DSA was repeated and showed aneurysm recanalization; it was promptly treated by endovascular approach with similar materials and coils deployment through the stent struts, achieving complete occlusion (Figure 2D). No antibiotics were necessary. The patient was discharged 5 days later using ASA, and remained asymptomatic during a 1-year follow-up. During that year, 2 attempts to perform image scan were made to document carotid patency and pseudo-aneurysm exclusion, but the patient missed both appointments.
Lateral radiography during endoscopy for treatment of esophageal restenosis: the esophagus is dilated with a balloon and a stent is deployed covering all atretic segment (A); Angiotomography with 3D reconstruction shows the fistulous tract (white arrow) from the proximal right carotid artery, just after the brachiocephalic trunk (double arrows) to the esophageal prosthesis (black arrow) (B); the stent is removed after endovascular treatment of the carotid esophageal fistula, and the endoscopy shows esophageal ulcer fully filled with coils (arrow) (C).
Angiogram of aorta (A) and right common carotid artery (B) shows carotid-esophageal fistula (arrows); the pseudoaneurysm is treated with stent and coils (C); after endoscopic removal of the esophageal stent (D), the recanalization is filled with additional coils (arrow).
Discussion
Anastomosis stricture is one of the most common complications of surgical repair of esophageal atresia, especially long-gap type as in our patient. 1 Endoscopic dilations usually have good results in esophageal stenosis, however, complex and tortuous stenosis may require an alternative therapeutic approach, 4 as seen in this case. Metallic stents have been preferentially used, instead of plastic ones, due to greater safety and easy deployment. 2 Their temporary use in benign conditions have good results, 5 however the time interval is still not well established, varying from 4 to 16 weeks, depending on the patient. 4 One complication may be a fistula between the esophagus and adjacent structures, which, despite its rarity, can occur even with large caliber vessels, such as the aorta, 6 subclavian artery 7 and the common carotid artery, 3 as in our case. Treatment options for cases of CEF include vascular surgery with vessel reconstruction, 8 artery ligation or endovascular treatment. 7,9 Vascular open surgery was not attempted in our case, considering the acute anemia associated with smaller blood volume in children. Endovascular treatment offers several options, such as conventional and covered stents, flow diverters, and embolization with detachable coils or liquid embolic agents. Covered stents represent a good option for ostium occlusion and, regardless of their successful use in high risk cases like ours, they were not available in our hospital. Thus, angioembolization was chosen because of its safety minimal invasiveness, and ability to control bleeding expeditiously and occlude the fistula. 10 Although arterial stenting alone is a well-stablished therapy in adults, its use in children remains controversial, due to the possibility of restriction of the arterial growth. Therefore, further long-term follow-up studies are needed, to better understand when to use these devices.
Conclusion
The CEF is a rare and serious complication of the metallic esophageal stents. Its early diagnosis and treatment require a high degree of suspicion, therefore reducing the associated morbidity and mortality rates.
Footnotes
Authors’ Note
Camila Girardi Fachin and Zeferino Demartini Jr contributed equally to the manuscript. The study was performed in accordance with the ICMJE Recommendations for the Protection of Research Participants and was approved by the local Ethics Committee. Informed written consent has been obtained from the patient’s legally authorized representative for the publication of the case report and accompanying images. All patient identification was removed to preserve anonymity. C.G.F., Z.D.J., B.C.A.T., C.B., E.A.B. and A.I.B.S.D.: conception of the work and acquisition of the data. C.G.F, Z.D.J., A.S.A.P and E.N.K.: analysis and interpretation of the data. C.G.F., Z.D.J., B.C.A.T. and E.A.B.: drafting of the work. A.S.A.P., E.N.K., C.B., and A.I.B.S.D.: critical revision of the work for intellectual content. C.G.F., Z.D.J., A.S.A.P., E.N.K., B.C.A.T., C.B., E.A.B. and A.I.B.S.D.: final approval of the version to be published. C.G.F., Z.D.J., A.S.A.P., E.N.K., B.C.A.T., C.B., E.A.B. and A.I.B.S.D.: responsibility for all aspects of the work ensuring that questions related to the accuracy or integrity of any part of the work is appropriately investigated and resolved.
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding
The author(s) received no financial support for the research, authorship, and/or publication of this article.
