Case report
Anaesthetic management of C-section in Brugada syndrome: When less is more
Manejo anestésico del síndrome de Brugada para cesárea: cuando menos es más
F. Marques da Costa
, M. Luís, F. Lança
Corresponding author
Serviço de Anestesiologia, Hospital de Santa Maria, Centro Hospitalar Lisboa Norte, Lisboa, Portugal
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Marques da Costa, M. Luís, F. Lança" "autores" => array:3 [ 0 => array:4 [ "nombre" => "F." "apellidos" => "Marques da Costa" "email" => array:1 [ 0 => "filipe.marquesdacosta@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "M." "apellidos" => "Luís" ] 2 => array:2 [ "nombre" => "F." "apellidos" => "Lança" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Serviço de Anestesiologia, Hospital de Santa Maria, Centro Hospitalar Lisboa Norte, Lisboa, Portugal" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Manejo anestésico del síndrome de Brugada para cesárea: cuando menos es más" ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Brugada syndrome (BrS) is a rare channelopathy. It was first identified in 1992, and is responsible for 4% of total sudden deaths.<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">1</span></a> The diagnosis includes typical electrocardiographic changes (coved-type ST segment elevation followed by an inverted T wave in right leads), possibly associated with other supraventricular arrhythmias or right bundle branch block.<a class="elsevierStyleCrossRefs" href="#bib0130"><span class="elsevierStyleSup">1–3</span></a> These are often triggered by physiological imbalance, such as intense physical activity, fever, or any other situation brought on by sympathetic stimulation. If symptomatic, the syndrome can manifest with syncope, palpitations, seizures, or even cardiac arrest.<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">1</span></a> Most diagnoses are reached during clinical investigation of the relatives of sudden death victims, since there is familiar transmission of BrS, with an autosomal dominant pattern but variable penetrance.<a class="elsevierStyleCrossRef" href="#bib0145"><span class="elsevierStyleSup">4</span></a> Some genetic disorders have been associated with BrS, SCN5A mutation being the most frequent. Patients usually manifest cardiac sodium channel defects, affecting the influx of sodium during depolarization; in other cases, potassium and calcium channel defects can also compromise repolarization.<a class="elsevierStyleCrossRef" href="#bib0150"><span class="elsevierStyleSup">5</span></a> Cardiac arrest occurs with ventricular fibrillation. Therefore, the treatment for symptomatic or high-risk patients is an implantable cardiodefibrillator (ICD).<a class="elsevierStyleCrossRefs" href="#bib0130"><span class="elsevierStyleSup">1,4</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Given its unpredictability, BrS is a true challenge for the anaesthesiologist. Local anaesthetics, which are commonly used in anaesthesia and are actually antiarrhythmics (class Ib), are amongst the potentially arrhythmogenic drugs for this condition.<a class="elsevierStyleCrossRefs" href="#bib0140"><span class="elsevierStyleSup">3,4,6</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">The following clinical case aims to expose a rare situation of a parturient previously diagnosed with high risk BrS in whom labour was induced at 37 weeks of pregnancy. Because of the familial transmission of BrS, the clinical challenge automatically involved the newborn, with early involvement of a cardiologist and neonatologist.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case-report</span><p id="par0020" class="elsevierStylePara elsevierViewall">A 37 year old pregnant woman (72<span class="elsevierStyleHsp" style=""></span>kg, 172<span class="elsevierStyleHsp" style=""></span>cm), American Society of Anesthesiologists (ASA) physical status III, was admitted to the delivery room at 37 weeks of pregnancy due to foetal blood flow redistribution (FBFR). She had been diagnosed with BrS type 1 two years previously (positive SCN5A mutation), with symptoms that led to implantation of an ICD, and had no history of cardiogenic shock or cardiac arrests since then. Her direct relatives (mother and brother) also had a confirmed diagnosis of BrS, with ICD placement. The patient had attended a high-risk-pregnancy evaluation with the anaesthesiologist at 35 weeks<span class="elsevierStyleHsp" style=""></span>+<span class="elsevierStyleHsp" style=""></span>6 days of pregnancy, in our hospital. The pregnancy was uneventful until 36 weeks<span class="elsevierStyleHsp" style=""></span>+<span class="elsevierStyleHsp" style=""></span>6 days, when she was admitted for obstetric monitoring due to FBFR. The electrocardiogram at admission was normal. Labour induction was then attempted with prostaglandin E2, with no response over the following 24<span class="elsevierStyleHsp" style=""></span>h. The patient's mild pain up to this point was easily managed with intravenous (IV) paracetamol. At this point, the obstetric team decided to proceed with a C-section. The patient was transferred to the operating room, monitored according to ASA standard recommendations, and the surgical safety checklist was performed. The ICD was deactivated, preventing electro-catheter interference, and was replaced with multifunction electrode pads connected to a manual defibrillator.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The anaesthetic protocol focused on pain and vagal blockade, as well as body temperature control. Regional anaesthesia with combined spinal epidural (CSE) was administered without complications. With the patient in a sitting position, an 18<span class="elsevierStyleHsp" style=""></span>G Touhy needle was inserted between L3 and L4, and contacted the epidural space at a depth of 5<span class="elsevierStyleHsp" style=""></span>cm. The dura mater was punctured with a 27<span class="elsevierStyleHsp" style=""></span>G pencil-point needle, injecting 1.8<span class="elsevierStyleHsp" style=""></span>mL of ropivacaine 0.75% (13.5<span class="elsevierStyleHsp" style=""></span>mg)<span class="elsevierStyleHsp" style=""></span>+<span class="elsevierStyleHsp" style=""></span>2.5 mcg sufentanil in the subarachnoid space. Sensory blockade up to T8 was obtained. The subarachnoid approach allowed us to administer lower doses of local anaesthesia, which minimized systemic absorption (compared to the epidural approach), thus ensuring less cumulative effect, lower plasma concentration, and therefore, less likelihood of cardiac toxicity. To minimize sympathetic blockade after CSE, the patient was lateralized to the left, avoiding aorto-caval compression and optimizing venous return. Rapid infusion of co-load with 500<span class="elsevierStyleHsp" style=""></span>mL of Ringer's lactate was performed. However, the patient presented brief episodes of hypotension (MAP<span class="elsevierStyleHsp" style=""></span><<span class="elsevierStyleHsp" style=""></span>65<span class="elsevierStyleHsp" style=""></span>mmHg), which were rapidly resolved with 5<span class="elsevierStyleHsp" style=""></span>mg boluses of IV ephedrine (25<span class="elsevierStyleHsp" style=""></span>mg in total). It was decided not to administer aspiration pneumonia prophylaxis, since the drugs available (metoclopramide and antihistamines) are contraindicated in BrS.<a class="elsevierStyleCrossRefs" href="#bib0140"><span class="elsevierStyleSup">3,7,8</span></a> The risk of aspiration was also minimized with the regional anaesthetic technique.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Surgery was uneventful, and the patient remained haemodynamically stable and in sinus rhythm (HR 55–90<span class="elsevierStyleHsp" style=""></span>bpm, MAP 60–80<span class="elsevierStyleHsp" style=""></span>mmHg) throughout the procedure. No arrhythmias or conduction disturbances were observed. Blood loss was approximately 400<span class="elsevierStyleHsp" style=""></span>mL. A total of 1000<span class="elsevierStyleHsp" style=""></span>mL of Ringer's lactate was infused. Intermittent boluses of IV propofol 30<span class="elsevierStyleHsp" style=""></span>mg, up to a total of 200<span class="elsevierStyleHsp" style=""></span>mg, were given for anxiolysis and sedation, particularly during the most vagotonic procedures such as foetus extraction, uterine eversion for suture, and placenta expulsion. The surgery lasted about 45<span class="elsevierStyleHsp" style=""></span>min and, as expected, there was no need for further anaesthesia through the epidural catheter, which was left in place for post-operative analgesia.</p><p id="par0035" class="elsevierStylePara elsevierViewall">A female newborn weighing 2780<span class="elsevierStyleHsp" style=""></span>g, with Apgar 9/10, was delivered without complications. After umbilical cord clamping, oxytocin infusion was started (10<span class="elsevierStyleHsp" style=""></span>U in 500<span class="elsevierStyleHsp" style=""></span>mL of normal saline over 1<span class="elsevierStyleHsp" style=""></span>h). The ICD was reactivated in the early post-operative period. Post-operative analgesia was achieved with 1<span class="elsevierStyleHsp" style=""></span>g of IV paracetamol plus 10<span class="elsevierStyleHsp" style=""></span>mg of IV ketorolac every 8<span class="elsevierStyleHsp" style=""></span>h, and 1<span class="elsevierStyleHsp" style=""></span>mg of epidural morphine every 12<span class="elsevierStyleHsp" style=""></span>h over the first 24<span class="elsevierStyleHsp" style=""></span>h. Four mg of IV ondansetron were given for nausea and vomiting prophylaxis. Continuous electrocardiographic monitoring was maintained during the first 24 postoperative hours in order to detect arrhythmias as soon as possible. No complications were observed during the patient's hospital stay, and she was discharged at day 4.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0040" class="elsevierStylePara elsevierViewall">Given its rarity, there are few reports of anaesthetics management in pregnant patients with BrS.<a class="elsevierStyleCrossRefs" href="#bib0135"><span class="elsevierStyleSup">2,9,10</span></a> Most receive general anaesthesia (GA).<a class="elsevierStyleCrossRefs" href="#bib0180"><span class="elsevierStyleSup">11–15</span></a> This however, particularly in obstetric patients, has its limitations: it requires anticipated difficult airway management,<a class="elsevierStyleCrossRef" href="#bib0205"><span class="elsevierStyleSup">16</span></a> the use of IV and inhalational anaesthetics might be harmful to the newborn by disturbing placental circulation and leading to oxygenation disorders and respiratory depression,<a class="elsevierStyleCrossRefs" href="#bib0210"><span class="elsevierStyleSup">17,18</span></a> and it can also trigger malignant arrhythmias in patients with BrS.<a class="elsevierStyleCrossRef" href="#bib0140"><span class="elsevierStyleSup">3</span></a> Regional anaesthesia (RA), which avoids these complications, is widely used in obstetric analgesia. Furthermore, the primary mechanism of action of the local anaesthetics required in RA is sodium channel blockade, which blocks the action potential. Local anaesthetics are therefore considered antiarrhythmic drugs (class Ib). However, in patients with SCN5A mutation, such as BrS, they are in fact potentially arrhythmogenic.<a class="elsevierStyleCrossRefs" href="#bib0140"><span class="elsevierStyleSup">3,19</span></a> This is particularly important when considering epidural blockade, which involves the administration of large volumes of local anaesthetic, leading to higher total plasma concentrations. This is based on evidence of the lack of metabolism in the epidural or subarachnoid space, suggesting that the plasma levels of local anaesthetic following neuraxial blockade are directly related to the total neuraxial dose administered. Similarly, local anaesthetic from the epidural space is absorbed in 2 stages (due to extensive vascularity and the presence of epidural fat), with rapid initial absorption followed by a slower second phase. This, therefore, would present a risk in both the intraoperative and post-operative period, particularly with prolonged infusions or repeated boluses.<a class="elsevierStyleCrossRef" href="#bib0225"><span class="elsevierStyleSup">20</span></a> The subarachnoid approach, therefore, is preferred in these patients, due to minimal systemic absorption and the need for small doses to achieve blockade.<a class="elsevierStyleCrossRefs" href="#bib0225"><span class="elsevierStyleSup">20,21</span></a> In terms of the choice of local anaesthetic, ropivacaine is less cardiotoxic than bupivacaine, which exhibits a “slow in, slow out” effect, due to higher cardiac sodium channel affinity.<a class="elsevierStyleCrossRefs" href="#bib0155"><span class="elsevierStyleSup">6,22,23</span></a> Control of anxiety and vagal reaction is as important as anaesthetic and analgesic management, since they are known to trigger ventricular tachycardia/fibrillation in patients with BrS.<a class="elsevierStyleCrossRef" href="#bib0140"><span class="elsevierStyleSup">3</span></a> In this case, propofol was used, since there is ample evidence of its safety in the literature.<a class="elsevierStyleCrossRefs" href="#bib0180"><span class="elsevierStyleSup">11–15</span></a> This particular drug has pharmacokinetic and pharmacodynamic advantages, such as rapid onset and short duration of action, particularly when compared with other sedatives such as benzodiazepines. Knowledge of the key moments of surgery (delivery, placenta expulsion, and uterine eversion) and careful coordination with the obstetrician are crucial to prevent unnecessary discomfort and pain.<a class="elsevierStyleCrossRef" href="#bib0245"><span class="elsevierStyleSup">24</span></a> In our case, we preferred intermittent, low-dose propofol boluses over continuous infusion, because sedation requirements fluctuated during surgery: there were periods where the patient was fully awake and bonding with the newborn, and other more vagotonic periods in which discomfort was managed with mild, brief sedation. In the post-operative period, we prioritized close electrocardiographic monitoring for at least 24<span class="elsevierStyleHsp" style=""></span>h, ICD reactivation,<a class="elsevierStyleCrossRef" href="#bib0140"><span class="elsevierStyleSup">3</span></a> and minimization of arrhythmia triggers, preferably avoiding local anaesthetics and opioids such as tramadol.<a class="elsevierStyleCrossRef" href="#bib0140"><span class="elsevierStyleSup">3</span></a> Ondansetron was chosen for nausea prophylaxis because previous reports and case series showed that it is safe in Brugada patients.<a class="elsevierStyleCrossRefs" href="#bib0140"><span class="elsevierStyleSup">3,4,25</span></a> However, because of its potential to prolong the QT interval, we suggest using it with caution and close monitoring. Body temperature must be monitored, using antipyretics such as paracetamol and non-steroidal anti-inflammatory drugs, if necessary.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusion</span><p id="par0045" class="elsevierStylePara elsevierViewall">We report this case in order to stimulate a debate on the use of RA in BrS patients. RA already offers numerous advantages over GA in the obstetric population, insofar as it does not require airway management, and has minimal repercussions on the newborn. However, local anaesthetics are arrhythmogenic in BrS. Our protocol permitted the use of RA and minimized the risk of malignant events, suggesting that a subarachnoid approach with low-dose local anaesthetic might be a safe alternative in this specific population. The whole peri-partum period requires careful planning, starting with a pre-anaesthesia evaluation to establish trust with the patient and calm their anxiety. We believe that a multidisciplinary approach is essential, referring complex patients to hospitals that can provide differentiated care.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Funding</span><p id="par0050" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Conflict of interest</span><p id="par0055" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:11 [ 0 => array:3 [ "identificador" => "xres1196202" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1114600" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1196203" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1114601" "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" => "Conclusion" ] 8 => array:2 [ "identificador" => "sec0025" "titulo" => "Funding" ] 9 => array:2 [ "identificador" => "sec0030" "titulo" => "Conflict of interest" ] 10 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2018-09-14" "fechaAceptado" => "2019-01-21" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1114600" "palabras" => array:3 [ 0 => "Brugada syndrome" 1 => "Arrhythmia" 2 => "Subarachnoid anestesia" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1114601" "palabras" => array:3 [ 0 => "Síndrome de Brugada" 1 => "Arritmia" 2 => "Anestesia subaracnoidea" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Brugada syndrome (BrS) is a rare and high risk condition, seldom encountered in the delivery room. Pregnant patients with BrS benefit from the lowest possible doses of arrhythmogenic drugs such as local anaesthetics. Based on this premise, the following case report exposes how a subarachnoid approach might be a desirable technique in C-section procedures for BrS patients. Pain and anxiety management are priorities in this specific population. Peri-partum planning, with a previous anesthesiology appointment and mutidisciplinary care in tertiary hospital facilities are crucial to avoid complications.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">El síndrome de Brugada (SBr) es una condición infrecuente y de alto riesgo que rara vez se encuentra en la sala de partos. Las pacientes embarazadas con SBr se benefician de las dosis más bajas posibles de fármacos arritmogénicos, como los anestésicos locales. Sobre la base de esta premisa, el siguiente caso expone cómo un abordaje subaracnoideo podría ser una técnica deseable en los procedimientos de cesárea para pacientes con SBr. El manejo del dolor y la ansiedad es prioritario en esta población específica. La planificación periparto, con una cita previa de anestesiología, y la atención multidisciplinaria en las instalaciones de hospitales terciarios son cruciales para evitar complicaciones.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Marques da Costa F, Luís M, Lança F. Manejo anestésico del síndrome de Brugada para cesárea: cuando menos es más. 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