Case report
Massive right hemothorax due to idiopathic spontaneous rupture of a phrenic artery following cardiac surgery
Hemotórax masivo derecho por rotura espontánea idiopática de una arteria frénica tras cirugía cardiaca
M. Taboada Muñiza,
, A. Tubio Posea, E. Ferreiroa Mosqueraa, A. Calvo Reya, J.M. Martínez Cereijob, J. Alvarez Escuderoa
Corresponding author
a Unidad de Cuidados Críticos Postoperatorios, Servicio de Anestesia, Reanimación y Tratamiento del Dolor, Hospital Clínico Universitario de Santiago, Santiago de Compostela, Galicia, Spain
b Servicio de Cirugía Cardiaca, Hospital Clínico Universitario de Santiago, Santiago de Compostela, Galicia, Spain
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Taboada Muñiz, A. Tubio Pose, E. Ferreiroa Mosquera, A. Calvo Rey, J.M. Martínez Cereijo, J. Alvarez Escudero" "autores" => array:6 [ 0 => array:4 [ "nombre" => "M." "apellidos" => "Taboada Muñiz" "email" => array:1 [ 0 => "manutabo@yahoo.es" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "A." "apellidos" => "Tubio Pose" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "E." "apellidos" => "Ferreiroa Mosquera" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "A." "apellidos" => "Calvo Rey" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 4 => array:3 [ "nombre" => "J.M." "apellidos" => "Martínez Cereijo" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 5 => array:3 [ "nombre" => "J." "apellidos" => "Alvarez Escudero" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Unidad de Cuidados Críticos Postoperatorios, Servicio de Anestesia, Reanimación y Tratamiento del Dolor, Hospital Clínico Universitario de Santiago, Santiago de Compostela, Galicia, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Cirugía Cardiaca, Hospital Clínico Universitario de Santiago, Santiago de Compostela, Galicia, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Hemotórax masivo derecho por rotura espontánea idiopática de una arteria frénica tras cirugía cardiaca" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1271 "Ancho" => 2333 "Tamanyo" => 267514 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Contrast-enhanced computed tomography. Active intrathoracic bleeding in the right hemithorax (white arrow) with significant haemothorax and clots that displaced liver and right kidney.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Haemothorax caused by spontaneous rupture of a phrenic artery is a very rare entity that requires prompt diagnosis and treatment to avoid hypovolemic shock and death. In most cases published so far, rupture of a phrenic artery is usually associated with blunt chest trauma.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">1,2</span></a> We present the case of a patient with late postoperative spontaneous rupture of the right phrenic artery with massive haemothorax and hypovolemic shock after cardiac surgery.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case report</span><p id="par0010" class="elsevierStylePara elsevierViewall">The patient was a 35-year-old man with no medical history of interest, admitted for endocarditis on a bicuspid aortic valve complicated by aortic vein perforation and severe aortic insufficiency. The patient underwent a metallic aortic valve replacement (carbamedics number 25). The surgery was uneventful. The patient was extubated after 6<span class="elsevierStyleHsp" style=""></span>h and discharged to the hospital ward 24<span class="elsevierStyleHsp" style=""></span>h after surgery. Anticoagulation was started with 60<span class="elsevierStyleHsp" style=""></span>mg enoxaparin/12<span class="elsevierStyleHsp" style=""></span>h and low-dose acenocumarine. On the seventh postoperative day, while on the ward, the patient and reported severe right chest pain radiating to the axila and respiratory distress. Blood tests showed Hb: 9.8<span class="elsevierStyleHsp" style=""></span>g/dl, INR: 1.45<span class="elsevierStyleHsp" style=""></span>s, PT time: 1.46<span class="elsevierStyleHsp" style=""></span>s, and 500,000 platelets. The patient's blood pressure fell to 70/45<span class="elsevierStyleHsp" style=""></span>mmHg, with poor response to fluid replacement. Norepinephrine perfusion was started and the patient was admitted to the postoperative Critical Care Unit (CCU). Right femoral artery and right internal jugular vein lines were placed for monitoring, together with peripheral lines were placed for fluid replacement. On arrival at the CCU Hb was 4.6<span class="elsevierStyleHsp" style=""></span>g/dl. Transfusion of packed red blood cells and fresh plasma was started. The transthoracic echocardiogram ruled out cardiac tamponade, and massive right haemothorax was diagnosed after chest X-ray and chest ultrasound (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>a). The patient was intubated and a chest tube was immediately inserted in the right haemothorax, through which 1600<span class="elsevierStyleHsp" style=""></span>ml of blood were collected. The chest radiograph was repeated, observing the persistence of massive haemothorax (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>b). We decided to perform urgent contrast-enhanced computerised axial tomography, which showed active right-sided intrathoracic bleeding with major haemothorax and clots that displaced the liver and right kidney (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). The cardiac, thoracic and interventional radiology services were contacted, and in view of the patient's haemodynamic and respiratory instability, urgent surgical intervention was decided. A median sternotomy was performed, with no sign of complications in the aorta and inferior vena cava. The right pleural cavity was opened, and abundant clots and blood were drained from the right hemithorax. Active bleeding from a phrenic artery at the base of the right diaphragm was observed. The artery was ligated, and good haemostatic control was achieved. The surgery was completed and the patient was transferred to the CCU. The patient received in total 10 bags of packed red blood cells and 9 of fresh plasma. Postoperative progress was good enough to allow withdrawal of catecholamines and extubation at 8<span class="elsevierStyleHsp" style=""></span>h after surgery, followed by transfer to the ward 48<span class="elsevierStyleHsp" style=""></span>h after surgery.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0015" class="elsevierStylePara elsevierViewall">Haemothorax after cardiac surgery is a relatively frequent entity that is usually associated with bleeding caused by dissection of the mammary artery in patients undergoing coronary revascularisation; with damage to an intercostal artery during placement of an intrathoracic tube; or as a complication of central venous access. In this case, the patient presented massive haemothorax due to spontaneous rupture of a phrenic artery on the seventh postoperative day after cardiac surgery. Previous studies have described haemothorax due to rupture of a phrenic artery after blunt trauma.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">1,2</span></a> Rupture of intrathoracic vessels has also been associated with diseases such as Ehlers Danlos, Marfan or neurofibromatosis type 1, which weaken the arterial wall and lead to the formation of aneurysms.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> In the few reports of haemothorax caused by spontaneous rupture of intrathoracic vessels and not by trauma or associated disease, bleeding was due to rupture of an intercostal artery.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a> Barau et al.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a> recently described a case of spontaneous haemothorax due to rupture of a phrenic artery 30<span class="elsevierStyleHsp" style=""></span>h after cardiac surgery. The authors believe that several factors could have contributed to the rupture, such as the administration of anticoagulants during cardiopulmonary bypass, or the shearing of pleural adhesions. Spontaneous haemothorax is sometimes preceded by spontaneous pneumothorax that causes the shearing of adhesions between the parietal and the visceral pleura.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> In our patient, the rupture of the phrenic artery occurred 7 days after the surgery. Anticoagulation with acenocoumarin, which was started on the third day despite low INR and PT, could have contributed to the magnitude of the haemothorax. The mechanism by which the phrenic artery is injured is unclear; however, it has been reported that spontaneous rupture of intrathoracic vessels may be associated with sustained hypertension, atelectasis, pulmonary infections, strenuous pulmonary physiotherapy or violent coughing, particularly in anticoagulated patients. We do not believe that any of these factors contributed to spontaneous rupture of the phrenic artery in our patient. Jang et al.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> described a case of spontaneous rupture of an intercostal artery in a patient with no history of trauma or illness, but with severe cough lasting several days. The ruptured artery was treated with arteriography and embolisation. Although transcatheter arterial embolisation could be an effective approach, in our case we decided to perform sternotomy to control the bleeding and drain the abundant thoracic clots due to the patient's severe haemodynamic and respiratory instability. To conclude, spontaneous rupture of a phrenic artery is extremely rare, but can lead to massive haemothorax and hypovolaemic shock. Early diagnosis and prompt treatment can be life-saving. The presence of sudden spontaneous chest pain associated with respiratory distress and haemodynamic deterioration is a warning sign of this rare, but not unknown, complication. Emergency CT, or better still, a CT angiogram, can be of great help in locating the origin of the bleed and will rule out the presence of vascular malformations.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflicts of interest</span><p id="par0020" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:3 [ "identificador" => "xres1024785" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec982679" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1024786" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec982680" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Case report" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec0020" "titulo" => "Conflicts of interest" ] 8 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2017-10-24" "fechaAceptado" => "2017-12-12" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec982679" "palabras" => array:3 [ 0 => "Phrenic artery" 1 => "Haemothorax" 2 => "Cardiac surgery" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec982680" "palabras" => array:3 [ 0 => "Arteria frénica" 1 => "Hemotórax" 2 => "Cirugía cardiaca" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">We report a case of a 35-years-old man who presented a massive haemothorax and hypovolemic shock following cardiac surgery, from spontaneous rupture of a phrenic artery. A quick diagnosis and immediate intervention is crucial to manage the patient.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Presentamos el caso de un hombre de 35 años que en el postoperatorio de cirugía cardiaca presenta un hemotórax masivo y shock hipovolémico por rotura espontánea de una arteria frénica. Un rápido diagnóstico y una inmediata intervención son cruciales para el manejo del paciente.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Taboada Muñiz M, Tubio Pose A, Ferreiroa Mosquera E, Calvo Rey A, Martínez Cereijo JM, Alvarez Escudero J. Hemotórax masivo derecho por rotura espontánea idiopática de una arteria frénica tras cirugía cardiaca. Rev Esp Anestesiol Reanim. 2018;65:291–293.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1056 "Ancho" => 2333 "Tamanyo" => 178840 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">(a) Chest X-ray showing massive haemothorax. (b) Chest X-ray showing persistence of haemothorax despite insertion of a chest tube (black arrows) and drainage of 1600<span class="elsevierStyleHsp" style=""></span>ml.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1271 "Ancho" => 2333 "Tamanyo" => 267514 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Contrast-enhanced computed tomography. Active intrathoracic bleeding in the right hemithorax (white arrow) with significant haemothorax and clots that displaced liver and right kidney.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:6 [ 0 => array:3 [ "identificador" => "bib0035" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Report of a rare case: occult hemothorax due to blunt trauma without obvious injury to other organs" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "F. Ogawa" 1 => "M. Naito" 2 => "A. Iyoda" 3 => "Y. 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