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Carballo Fernández, J. Arca Suárez, A. Prado Rodríguez, E. Freire Vila, D. Ruanova Seijo, M. Núñez Centeno" "autores" => array:6 [ 0 => array:4 [ "nombre" => "J." "apellidos" => "Carballo Fernández" "email" => array:1 [ 0 => "jesus.carballo.fernandez@sergas.es" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "J." "apellidos" => "Arca Suárez" ] 2 => array:2 [ "nombre" => "A." "apellidos" => "Prado Rodríguez" ] 3 => array:2 [ "nombre" => "E." "apellidos" => "Freire Vila" ] 4 => array:2 [ "nombre" => "D." "apellidos" => "Ruanova Seijo" ] 5 => array:2 [ "nombre" => "M." "apellidos" => "Núñez Centeno" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Anestesiología, Reanimación y Tratamiento del Dolor, Complejo Hospitalario Universitario A Coruña, A Coruña, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Hemorragia masiva tras fístula arterio-esofágica por una arteria subclavia aberrante desconocida" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 453 "Ancho" => 800 "Tamanyo" => 40023 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Angio-CT scan showing retroesophageal ARSA (arrow). Intra-oesophageal Sengstaken-Blakemore balloon.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Aberrant right subclavian artery (ARSA) is one of the most common congenital anomalies of the aortic arch, with an incidence of .5%–1% in the population. Its location is usually retroesophageal, and in most cases it is asymptomatic<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–4</span></a>. In patients with predisposing risk factors, it can lead to the formation of an arterio-oesophageal fistula after placement of an endotracheal tube, a nasogastric tube, or a salivary bypass tube in the oesophageal position over a long time, in addition to the effect of radiotherapy and adjuvant chemotherapy<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5–7</span></a>.</p><p id="par0010" class="elsevierStylePara elsevierViewall">We describe the case of a patient with a tracheo-oesophageal fistula after ENT surgery, who developed massive bleeding from an arterio-oesophageal fistula secondary to an unknown ARSA.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinical case</span><p id="par0015" class="elsevierStylePara elsevierViewall">The patient was a 50-year-old male with a history of smoking 40 cigarettes/day, drinking, addicted to parenteral drugs, with a body mass index of 19. The patient was diagnosed with an infiltrative squamous cell carcinoma of the left hypopharynx, treated by total laryngectomy, bilateral functional neck dissection, and placement of a phonatory prosthesis, with adjuvant radiotherapy and chemotherapy.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Seventeen months after the operation, the patient underwent closure of a pharyngo-tracheal fistula secondary to the initial surgery by means of a left deltopectoral flap, he was reoperated 2 months later for closure of a pharyngostoma (second stage, by rotation of the previous deltopectoral flap), made good progression and was discharged from resuscitation at 24<span class="elsevierStyleHsp" style=""></span>h.</p><p id="par0025" class="elsevierStylePara elsevierViewall">At 48<span class="elsevierStyleHsp" style=""></span>h, the patient presented on the ward with syncope and orotracheal bleeding, and was transferred to the operating theatre for urgent examination. He arrived in an obnubilated state, with cutaneous-mucosal pallor, hypotensive (mean arterial pressure of 49<span class="elsevierStyleHsp" style=""></span>mmHg), tachycardic, and with profuse bleeding from the mouth and tracheostoma. General anaesthesia was induced with fentanyl 150<span class="elsevierStyleHsp" style=""></span>μg, etomidate 15<span class="elsevierStyleHsp" style=""></span>mg, and rocuronium 60<span class="elsevierStyleHsp" style=""></span>mg, with doses adjusted to the patient's weight, and intubation through the tracheostoma. He was monitored invasively, requiring phenylephrine and noradrenaline to maintain mean arterial pressure >65<span class="elsevierStyleHsp" style=""></span>mmHg and, after the first arterial blood gas (haemoglobin 5<span class="elsevierStyleHsp" style=""></span>g/dl, haematocrit 17%), received intraoperative polytransfusion of 6 units of packed red cells, 500<span class="elsevierStyleHsp" style=""></span>ml of fresh frozen plasma, 1 pool of platelets, and 4<span class="elsevierStyleHsp" style=""></span>g of fibrinogen. The orotracheal area was checked surgically, and an upper gastrointestinal endoscopy was performed without clear visualisation of the responsible blood vessel, placing 3 haemostatic clips on the oesophageal wall 5<span class="elsevierStyleHsp" style=""></span>cm from the entrance of the oesophagus into the chest cavity, but it was not possible to pinpoint a main bleeding point. After an estimated blood loss of 2000<span class="elsevierStyleHsp" style=""></span>ml, the patient was haemodynamically stabilised and transferred to the resuscitation unit, sedated, and on mechanical ventilation. A thromboelastogram was performed, after which a further 500<span class="elsevierStyleHsp" style=""></span>ml of fresh frozen plasma was transfused, and arterial blood gas analysis showed haemoglobin levels of 8.3<span class="elsevierStyleHsp" style=""></span>g/dl and haematocrit of 28%.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Thirty minutes after arrival in resuscitation the patient had a further episode of massive haemorrhage, with significant haemodynamic instability, requiring a total transfusion, guided by thromboelastography, of another 6 units of packed red blood cells, 1000<span class="elsevierStyleHsp" style=""></span>ml of fresh frozen plasma, 2 platelet pools, and 3<span class="elsevierStyleHsp" style=""></span>g of fibrinogen, in addition to pharmacological support with boluses of intravenous adrenaline due to sudden hypotension and extreme bradycardia despite continuous perfusion of noradrenaline to maintain mean arterial pressure >65<span class="elsevierStyleHsp" style=""></span>mmHg. After an estimated blood loss of 1500<span class="elsevierStyleHsp" style=""></span>ml, control of the bleeding point was achieved by digital compression through the tracheostomy, and a Foley urinary catheter was temporarily placed by ENT in the resuscitation unit, after which the bleeding stopped and vasopressor support was withdrawn.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Once the patient was stabilised, the case was discussed with general surgery and it was decided to place a Sengstaken-Blakemore balloon in an unusual position, cranial to its usual location, in the upper third of the oesophagus and without inflating the distal balloon (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0040" class="elsevierStylePara elsevierViewall">Considering the patient's history of neck/chest radiotherapy, pharyngo-tracheal fistula due to radionecrosis, nasogastric tube, and salivary tube in oesophageal position for weeks, as well as the absence of oesophageal varices or visible vessels, it was decided to complete the study with an urgent angio-CT scan due to a suspected diagnosis of an aorto-oesophageal fistula, which revealed a previously unknown ARSA, with a retro-oesophageal course and a fistula between that artery and the oesophagus (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0045" class="elsevierStylePara elsevierViewall">To treat the patient, an endoluminal prosthesis consisting of a self-expanding nitinol stent covered on its inner side by polytetrafluoroethylene (Viabahn® prosthesis, Gore, Arizona, USA) was implanted in the vascular radiology department at the level of the right subclavian artery from its origin, with no subsequent bleeding.</p><p id="par0050" class="elsevierStylePara elsevierViewall">After a Sengstaken-Blakemore balloon for haemostatic control had been in situ for 24<span class="elsevierStyleHsp" style=""></span>h, it was deflated in the resuscitation room and an endoscopic revision was performed, with no evidence of bleeding points. A new nasogastric feeding tube was placed and a tracheostomy tube was placed, which is usual in postoperative care following this type of intervention.</p><p id="par0055" class="elsevierStylePara elsevierViewall">The patient remained in resuscitation for the following 48<span class="elsevierStyleHsp" style=""></span>h with serial analytical controls, remaining stable, and it was therefore decided to discharge him to the ward for further care. Given his good progress, he was discharged from the hospital 2 days later.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0060" class="elsevierStylePara elsevierViewall">ARSA is one of the most common congenital anomalies of the aortic arch, with the subclavian artery arising directly from the aorta with absence of the brachiocephalic trunk<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a>. This malformation may be associated with others, such as aneurysm of the origin of the descending thoracic aorta or Kommerell's diverticulum. ARSA has an incidence of approximately .5%–1% and it can be as high as 2.5%. Aberrant left subclavian artery is less common, with an incidence of .05%<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a>.</p><p id="par0065" class="elsevierStylePara elsevierViewall">In 80% of cases the ARSA passes posterior to the oesophagus, in 15% between the oesophagus and the trachea, and 5% anterior to the trachea<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,3,4</span></a>. It is usually asymptomatic, but when it does cause symptoms, the most common is dysphagia caused by oesophageal compression due to the retro-oesophageal location of the vessel. Other symptoms in order of frequency are: dyspnoea (retrotracheal ARSA), stridor, chest pain, cough, bronchial aspiration, recurrent lung infections, back pain, and numbness of the right upper limb<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,3,4</span></a>.</p><p id="par0070" class="elsevierStylePara elsevierViewall">A priori treatment is only recommended in ARSA with symptoms and/or if the diameter of the artery is greater than 3<span class="elsevierStyleHsp" style=""></span>cm, or the diameter of Kommerell's diverticulum is greater than 5.5<span class="elsevierStyleHsp" style=""></span>cm, due to the risk of rupture and dissection<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a>.</p><p id="par0075" class="elsevierStylePara elsevierViewall">Retro-oesophageal ARSA is particularly susceptible to extrinsic compression and pressure necrosis caused by semi-rigid devices such as a nasogastric or endotracheal tube, predisposing to the formation of an arterio-oesophageal fistula<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4–6</span></a>. Previous surgeries, infections, neoplasms, foreign bodies, radiotherapy, prolonged mechanical ventilation, trauma, previous laryngectomy, or previous oesophagectomy are risk factors for developing these fistulas<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7–9</span></a>.</p><p id="par0080" class="elsevierStylePara elsevierViewall">The consequence of the formation of an oesophageal-arterial fistula will be massive acute gastrointestinal bleeding that can lead to death of the patient in most of these situations<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,8–10</span></a>. In the event of haemorrhage presenting as bright red haematemesis, a fistula between the ARSA and the oesophagus should be suspected, requiring intensive resuscitation and intra-oesophageal tamponade with a Sengstaken-Blakemore balloon for initial management, stabilisation of the patient, and subsequent angio-CT, and/or arteriography to confirm or rule out the diagnosis<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">9,10</span></a>.</p><p id="par0085" class="elsevierStylePara elsevierViewall">In conclusion, the number of patients with risk factors for the development of an arterio-oesophageal fistula among the population undergoing oncological ENT or maxillofacial surgery is significant. In the case presented, previous surgery, subsequent radiotherapy, and chemotherapy, together with the presence of intra-oesophageal foreign bodies, led to the formation of this fistula, with an almost fatal outcome.</p><p id="par0090" class="elsevierStylePara elsevierViewall">We must not forget that around 1% of the population has this anatomical variant and that, given the risk factors that are so present in our resuscitation units, it is an anatomical disease to be taken into consideration in the event of major gastrointestinal bleeding to accelerate diagnosis and treatment.</p><p id="par0095" class="elsevierStylePara elsevierViewall">It is important to highlight the availability of rapid access to intra-oesophageal compression mechanisms such as the Sengstaken-Blakemore balloon, although, depending on the means available and in an emergency situation, a balloon urinary catheter (Foley type) can stop active bleeding, as demonstrated in this case, while awaiting a more definitive solution.</p><p id="par0100" class="elsevierStylePara elsevierViewall">However, it does not seem unreasonable to consider an angio-CT scan as a complement to the usual non-contrast CT scan in patients who are to undergo this type of surgery, with a high risk of fistula formation at the cervical level<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">11,12</span></a>. This would serve both to prevent certain procedures (such as avoiding instrumentation of the oesophagus), and to consider the possibility of major digestive bleeding in this region and act in a protocolised manner, thus reducing the morbidity and mortality of our patients.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Funding</span><p id="par0105" class="elsevierStylePara elsevierViewall">The authors declare that they have received no funding for this work.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Conflict of interests</span><p id="par0110" class="elsevierStylePara elsevierViewall">The authors have no conflict of interests to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:10 [ 0 => array:3 [ "identificador" => "xres1882249" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1631455" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1882248" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1631456" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Clinical case" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec0020" "titulo" => "Funding" ] 8 => array:2 [ "identificador" => "sec0025" "titulo" => "Conflict of interests" ] 9 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2021-08-19" "fechaAceptado" => "2021-10-30" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1631455" "palabras" => array:4 [ 0 => "Fistula" 1 => "Hemorrhage" 2 => "Subclavian artery" 3 => "Aberrant" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1631456" "palabras" => array:4 [ 0 => "Fistula" 1 => "Hemorragia" 2 => "Arteria subclavia" 3 => "Aberrante" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">The aberrant right subclavian artery has an incidence of 0.5%–1% in the population, generally with retroesophageal location. It can lead to the formation of an arterio-esophageal fistula in patients with predisposing risk factors due to devices placed in esophageal or tracheal position, as it is particularly susceptible to extrinsic compression and pressure necrosis.</p><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">We present the case of a patient with a postsurgical tracheoesophageal fistula, who developed massive bleeding due to an arterioesophageal fistula secondary to an unknown aberrant right subclavian artery. For hemostatic management, alternative maneuvers were performed, such as the placement of a Foley-type urinary catheter at the point of bleeding and the subsequent placement of a Sengstaken-Blakemore balloon in cranial position.</p><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Given the severity of the condition and the possible diagnostic delay, it seems appropriate to consider performing a preoperative CT angiography in patients with risk factors who undergo these procedures.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">La arteria subclavia derecha aberrante tiene una incidencia de un 0,5%−1% en la población, con una localización generalmente retroesofágica. Puede llevar a la formación de una fístula arterio-esofágica en pacientes con factores de riesgo predisponentes por dispositivos colocados en posición esofágica o traqueal, al ser particularmente susceptible a la compresión extrínseca y a la necrosis por presión.</p><p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Presentamos el caso de un paciente con una fístula tráqueo-esofágica postquirúrgica, que desarrolló un sangrado masivo por una fístula arterio-esofágica secundaria a una arteria subclavia derecha aberrante desconocida. Para el manejo hemostático se realizaron maniobras alternativas, como la colocación de una sonda urinaria tipo Foley en el punto de sangrado y la posterior colocación de un balón de Sengstaken-Blakemore en posición craneal.</p><p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">Dada la gravedad del cuadro y el posible retraso diagnóstico, parece adecuado considerar la realización de un angioTC preoperatorio en pacientes con factores de riesgo sometidos a estos procedimientos.</p></span>" ] ] "multimedia" => array:2 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 916 "Ancho" => 905 "Tamanyo" => 121315 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Sengstaken-Blakemore balloon in unusual position, in proximal oesophageal position without distal balloon inflation.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 453 "Ancho" => 800 "Tamanyo" => 40023 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Angio-CT scan showing retroesophageal ARSA (arrow). 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Case report
Massive hemorrhage after arterioesophageal fistula from an unknown aberrant subclavian artery
Hemorragia masiva tras fístula arterio-esofágica por una arteria subclavia aberrante desconocida
J. Carballo Fernández
, J. Arca Suárez, A. Prado Rodríguez, E. Freire Vila, D. Ruanova Seijo, M. Núñez Centeno
Corresponding author
Servicio de Anestesiología, Reanimación y Tratamiento del Dolor, Complejo Hospitalario Universitario A Coruña, A Coruña, Spain